292 Gut 1995; 37: 292-295 Small intestinal stricture complicating superior mesenteric vein thrombosis. A study of three cases Gut: first published as 10.1136/gut.37.2.292 on 1 August 1995. Downloaded from

C Eugene, D Valla, L Wesenfelder, A Fingerhut, A Bergue, J Merrer, C Felsenheld, A Moundji, J-C Etienne

Abstract Previous history included cocaine addiction, Mesenteric vein thrombosis associated which ended in 1984. There was no history of with intestinal stricture, as a consequence thrombosis in the family. Investigations per- of intestinal ischaemia, has only been formed in another hospital showed leucocytosis mentioned twice in published works. The whereas the pancreatic enzymes and hepatic clinical, biological, and morphological function tests were normal. Ultrasonography aspects as well as the treatment of this and computed tomography were not contribu- morbid association were studied in three tive. Upper gastrointestinal endoscopy showed patients. In all, a two stage clinical course a congestive gastropathy. Upon admission on (initial acute and fever, 25 November 1990, physical examination was followed by chronic intestinal obstruc- normal. Leucocytosis was still present. The tion), corresponding to the sequence head of the pancreas was found to be heteroge- thrombosis/stricture, was found. x Ray neous on endosonography, computed tomo- studies showed a regularly contoured graphy, and nuclear magnetic resonance. The intestinal stricture. Surgical resection was main pancreatic duct was found to be normal required in all three cases for stricture, on endoscopic retrograde choliopancreatic- associated in one case with mesenteric ography. The biliary duct was not opacified. infarction. Anticoagulation treatment was There was no erosive . Ultra- used to preclude recurrence. Increased sonography and duplex scanning showed clinical awareness could lead to the diag- thrombosis of the portal vein with cavemoma nosis of intestinal stricture secondary to and splenomegaly. Computed tomography and mesenteric vein thrombosis more often nuclear magnetic resonance showed that both and at an earlier stage. Treatment the portal and superior mesenteric veins were consists of evaluation of predisposing thrombosed, as confirmed by arteriograms, features, intestinal resection when neces- which showed hepatopetal flow in the caver- http://gut.bmj.com/ sary, and anticoagulation therapy, as noma and . Colonoscopy showed indicated. an adenoma. Haematological tests were nega- (Gut 1995; 37: 292-295) tive except for the presence of antiphospholipid antibody, but this abnormality was not found Keywords: mesenteric vascular occlusion, mesenteric ultimately (see Table II). Heparin therapy was veins, thrombosis, , small , intestine. started 1 December 1990. On 21 December, and the patient presented with proximal intestinal on September 28, 2021 by guest. Protected copyright. Unit obstruction. A barium follow through and C Eugene L Wesenfelder computed tomography were thought to be con- Mesenteric ischaemia resulting from portal or sistent with jejunal haematoma. Total paren- General and Digestive superior mesenteric venous thrombosis can be teral nutrition was started. As these symptoms Surgery Unit A Fingerhut seen in patients without hepatic disease or persisted and a second barium follow through A Moundji ongoing abdominal disease.' 2 Apparently showed a stricture (Fig 1), an exploratory J-C Etienne primitive venous thrombosis is most often laparotomy was performed on 25 January Pathology Department associated with patient or latent myeloprolifer- 1991. A jejunal stricture was found at 30 cm A Bergue ative syndromes or coagulation disorders.' 3-5 from the duodenojejunal flexure, associated C Felsenheld Mesenteric vein thrombosis can lead to sub- with adherence to the omentum, which was Medical Intensive Care acute reversible manifestations (such as severe, resected as well. Histopathological study (by Unit often mid-abdominal pain, occasionally resem- J Merrer bling abdominal , associated with or 3 6-8 or Centre Hospitalier without intestinal haemorrhage)2 Intercommunal, mesenteric infarction.9 The development of Poissy, France intestinal stricture has only been mentioned in two 6 We described the characteristics Hepatology and cases.2 Gastroenterology Unit, of intestinal stricture secondary to mesenteric Piti6 Salp6triere venous thrombosis based on our experience in Hospital, Paris, three cases. France D Valla Correspondence to: Professor C Eugene, Centre Case reports Hospitalier Intercommunal, 10 rue du Champ Gaillard, 78303 Poissy Cedex, France. Case report 1 Accepted for publication A 36 year old man was referred to our hospital Figure 1: Small intestinefollow through showing a long, 19 December 1994 in November 1990 for acute abdominal pain. regularly contoured stricture (case 1). Small intestinal stricture complicating superior mesenteric vein thrombosis. A study ofthree cases 293

intestines were not volvulated. Histopatho- ...... logical examination showed haemorrhagical infarction with numerous recent thrombosis of the veins in the submucosa and of larger

mesenteric veins. The patient was given Gut: first published as 10.1136/gut.37.2.292 on 1 August 1995. Downloaded from heparin postoperatively and made an unevent- ful recovery. Research for haematological disease was negative (Table II). Coumarin therapy was started. No relapse was seen at the last follow up visit in September 1994.

Case report 3 A 54 year old man was admitted to hospital in proximal dilatation (case 3). November 1991 for acute abdominal pain. There was a history of appendicectomy, hyper- I Bad Alsamad) showed a 4 cm ulceration, lipidaemia, angina pectoris, hypertension, and fibrosis disrupting the architecture of the mus- unexplained thrombosis of the radial artery in cular fibres, and thrombosis of the vessels with 1986. There was no history ofthrombosis in the mild to moderate recanalisation. There was no family. Drugs taken by the patient included: haematoma. The patient made an uneventful fenofibrate, nifedipin, and bisoprolol. Clinical recovery. Heparin therapy was stopped and findings included hypogastric pain, vomiting, replaced by coumarin. No relapse was seen at blood per anum, and fever (39'C). The the last follow up visit at the end of 1993. was tender to examination without guarding. The white blood count showed leuco- cytosis and transient hyperthrombocytaemia Case report 2 (570 000/,u). Serum lipase was 3.5 times A 46 year old man was admitted to hospital in normal, while amylase was normal. Ultra- September 1991 for acute abdominal pain. sonography, computed tomography, upper This patient had taken aspirin for a toothache gastrointestinal endoscopy, colonoscopy, and several days previously. There was no history of barium gastroduodenal follow through were thrombosis in the family. Periumbilical pain normal. As pain persisted, repeat ultrasono- was associated with dark red blood per anum. graphy was performed four months later and The abdomen was tender to examination but showed portal vein thrombosis. Intestinal without guarding. The white blood count angiograms showed thrombosis of the splenic showed neutrophil polymorphonucleosis. and mesenteric veins as well. The pancreas was http://gut.bmj.com/ Antral and bulbar erosions were found normal on computed tomography and upon upper gastrointestinal endoscopy and endosonography. Two months later he had disappeared after antisecretory treatment. abdominal pain consistent with Koenig's syn- Colonoscopy and ultrasonography were non- drome. A barium swallow with follow through contributive. As abdominal pain persisted, an showed a short jejunal stricture (2 cm), associ- exploratory laparotomy was performned on ated with proximal dilatation (Fig 2). Distended on September 28, 2021 by guest. Protected copyright. 12 November 199 1. A 2 cm stricture was found intestinal loops were found on laparotomy and on the , 20 cm from the valvule of Bauhin, thought to be caused by a stricture of ischaemic attached to the bladder by a band. Histo- origin. Distant from the stricture, a peritoneal pathological examination showed an ulceration band was found, which was resected along with with granulomatous inflammatory infiltrate, the stricture. Histopathological examination without visible thrombosis. In August 1992, showed a non-ulcerated stenosis with non- repeat laparotomy was performed for intestinal specific inflammatory infiltrate. In submucosa obstruction associated with guarding. A 30 cm and mesentere, vein walls were thickened by segment of infarcted ileum, thought to be caused by venous thrombosis, was resected. TABLE II Haematological investigations

There were no bands and the small and large Cases

TABLE i Abdominal investigations 1 2 3 Cases Bone marrow culture* N N N 1 2 3 Platelets N N Nt Protein S NN N US Heterogeneous pancreatic head N N Protein C N N N CT Heterogeneous pancreatic head N N AT III N N N MRI Heterogeneous pancreatic head ND ND PT, APT N N N Endosonography Heterogeneous pancreatic head N N Factors II, V, VII, and X N N N ERCP N N ND Fibrinogen N N N Gastroscopy Ulcerations* Ulcerations* Varix Plasminogen ND N N Colonoscopy 8 mm N N Euglobulin lysis ND N N Angiograms Nt ND Nt After venous occlusion ND NN Ham-Dacie test N ND N Amylasaemia N N N Circulating anticoagulants N N N Lipasaemia ND N 3-5 N Antiphospholipid antibodies +1 N N Laparotomy N N N Anticardiolipin antibodies N N N

US=ultrasonography; MRI=magnetic resonance imaging; CT=computed tomography; N: normal or negative result; *=without addition of ERCP=endoscopic retrograde cholangiography and wirsungography. *Duodenal erosions erythropoietin; t=transientiy increased; t-transiently positive; (case 1); antral and duodenal erosions (case 2); tnormal apart from the mesenteric thrombosis; AT III=antithrombin III; PT=prothrombin time; APT= N=normal or negative result; ND=not done. activated partial thromboplastin time; ND=not done. 294 Eugene, Valla, Wesenfelder, Fingerhut, Bergue, Merrer, Felsenheld, Moundji, Etienne

fibrosis, which contained neovascularisation. there has not been any further bowel disease. This pattern was consistent with previous Intestinal strictures, associated with mesen- venous thrombosis with secondary repoermabil- teric vein thrombosis, are probably the conse- isation but no recent thrombosis was seen. The quence of ischaemia because of the following

postoperative course was uneventful. Haemato- findings. Firstly, in our patients, a two stage Gut: first published as 10.1136/gut.37.2.292 on 1 August 1995. Downloaded from logical tests were negative (Table II). The sequence was found. Acute abdominal pain patient was treated longterm with coumarin in and fever, accompanied in two cases by blood December 1992. No relapse was seen at the last from the anum were the initial manifestations. follow up visit in September 1994. This corresponds to the initial ischaemia.2 3 6-8 Secondly, regularly contoured stricture is con- sistent with ischaemia. The roentgenoligical Discussion signs reported by Cardot et al2 and Witte et al,6 Mesenteric vein thrombosis is a rare disease. also suggest an ischaemic mechanism. Thirdly, Diagnosis, and the research of causative in our patients, histological examination con- factors, are difficult. Recognised promoting firmed the mucosal (ulcerations in two cases) factors include ,10 intra-abdominal and submucosal (fibrosis or oedema and or haematological causes,1 45 a patent or latent haemorrhage) involvement. Ischaemia is myeloproliferative syndrome,4 ll protein C12 13 further supported by the occlusion of neigh- or S,14 or antithrombin 1115 or plasminogen bouring vessels. activator16 deficiencies. In one case associated Surprisingly, strictures are rarely mentioned with intestinal stricture, thrombosis was sec- in reports of mesenteric vein thrombosis. The ondary to a deficit of the cofactor II or reason is not obvious. Two of our patients had heparin.2 Antiphospholipid antibodies were taken aspirin just before the initial manifesta- transiently noted in our case 1; no signs of sys- tions. By analogy with lesions attributed to temic lupus erythematosus were seen either at non-steroid anti-inflammatory drug treat- the time of admission or during follow up. ment,'9 it might be hypothesised that these These antibodies have been associated with drugs played a part in the constitution of the mesenteric vein thrombosis.17 This patient intestinal lesions. This should not obscure the (case 1) had ceased to be a drug addict, which fact, however, that the complete occlusion of can cause intestinal infarction without throm- the small mesenteric vessels is an indisputable bosis,18 long before the present episode of proof of primary vascular involvement. In all thrombosis occurred. our three cases, the surgeon noted a peritoneal Signs of chronic intestinal obstruction band in contact with the stricture, but the appeared several weeks after the presumed intestine was neither the site of or onset of mesenteric vein thrombosis in all strangulation. The implications of associated three cases, but the formal diagnosis of bands remain a mystery, but they might be http://gut.bmj.com/ intestinal obstruction was made only several related to subacute torsion, a possible cause of weeks to several months afterwards. In one mesenteric vein occlusion, as we have pre- patient receiving heparin, the diagnosis of viously reported.20 mural haematoma was wrongly considered. In The diagnosis ofmesenteric vein thrombosis the two patients for whom barium follow associated with intestinal stricture could prob- through studies were obtained, the image was ably be made more often and earlier if only the of a short (2 cm) or long (5 cm) regularly con- clinician were aware of its possibility. This on September 28, 2021 by guest. Protected copyright. toured stricture (Figs 1 and 2). Cardot et al 2 would lead to an aetiological inquiry, intestinal saw a moderately tight, 15 cm long stricture resection whenever necessary, and anticoagu- in the proximal intestine, with spiculed bor- lation, as indicated. 6 ders. Witte et al reported the acute onset of We would like to thank Catherine Collet, MD, Florence ascites and mesenteric and portal vein throm- Mathonnet, PhD, Thi Vu, MD (Poissy), and Marie-Helene Horellou, MD (Paris) for their assistance in the haematological bosis in a 71 year old man. Later, a barium investigations, Nicole Casadevall, MD (Garches) for the bone follow through showed oedema, spicules, marrow cultures and I Bad Alsamed, MD (Paris) for the pathol- thumb prints in the distal ileum. No patho- ogy study in case 1. logical data were provided in either of these 1 Grendell JM, Ockner RK. Mesenteric venous thrombosis. cases. Gastroenterology 1982; 82: 358-72. In our patients, the anatomical lesions were 2 Cardot F, Borg JY, Guedon C, Lerebours E, Colin R. Les syndromes d'ischemie veineuse mesenterique: infarctus et diversified, suggesting that they corresponded ischemie transitoire. Gastroenterol Clin Biol 1992; 16: to different stages of intestinal involvement. 644-8. Mucosal ulceration was found only in two 3 Harward TRS, Green D, Bergan JJ, Rizzo RJ, Yao JST. Mesenteric vein thrombosis. J Vasc Surg 1989; 9: patients operated on early after the onset of 328-33. 4 Valla D, Casadevall N, Huisse MG, Tulliez M, Grange JD, symptoms. Muller 0, et al. Etiology of portal venous thrombosis in Anticoagulation treatment was started adults. A prospective evaluation of primary myeloprolifer- ative disorders. Gastroenterology 1988; 94: 1063-9. because no curable cause of thrombosis could 5 Marzelle J. Thromboses portale et mesenterique. Etiologie, be found (Tables I and II). With anticoagula- manifestations cliniques et traitement. Ann Gastroenterol Hepatol 1990; 26: 51-60. tion treatment, all patients have been free of 6 Witte CL, Brewer ML, Witte MH, Pond GB. Protean recurrent thrombosis, suggesting the use of manifestations of pylethrombosis. A review of thirty-four patients. Ann Surg 1985; 86: 191-202. longterm anticoagulation treatment for this 7 Verbanck JJ, Rutgeerts U, Haerens MH, Tytgat JH, Segaert condition. MF, Tytgat HF, et al. Partial splenoportal and superior mesenteric venous thrombosis. Early sonographic diag- In all three patients, the stricture of the small nosis and successful conservative management. intestine developed during the post-throm- Gastroenterology 1984; 86: 949-52. 8 Clavien PA, Huber 0, Rohner A. Venous mesentenic botic course, and became symptomatic, ischemia: conservative or surgical treatment? Lancet 1989; requiring surgical resection. After surgery, ii: 48. Small intestinal stricture complicating superior mesenteric vein thrombosis. A study of three cases 295

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