preoperative intraoperative of ofobscure - gut · patient with blue rubber bleb naevus syndrome....

Gut, 1987, 28, 869-877

Preoperative and intraoperative localisation ofgastrointestinal bleeding of obscure originW Y LAU, S T FAN, S H WONG, K P WONG, G P POON, K W CHU,W C YIP, AND K K WONG

From the Government Surgical Unit, Queen Mary Hospital, Pokfulam Road, Hong Kong

SUMMARY In the past six years, 37 patients with gastrointestinal bleeding of obscure origin hadtheir bleeding sites localised preoperatively or intraoperatively. Preoperative investigationsfollowed a regime consisting of endoscopy, barium meal and follow through, small bowel enema,9mTc pertechnetate scan, 'mTc-labelled red blood cell scan and selective coeliac and mesentericangiography. Bleeding lesions were localised preoperatively in 36 patients. In one patient,diagnostic laparotomy had to be carried out immediately before any investigation because thebleeding was severe. At operation, angiosarcoma of ileum was found. Unless preoperativeinvestigations showed the lesions to be in anatomically fixed organs like the duodenum or colon,the lesions had still to be found at operation. Palpation and transillumination detected the lesionintraoperatively in 21 patients while only some lesions were found in three patients with multiplelesions. Sigmoidoscopy through enterotomies was required in one patient. Intraoperativeenteroscopy was done for small lesions not found grossly at operation in nine patients, to detectadditional lesions in three patients or to rule out suspicious lesion shown on preoperative tests inone patient. In another patient with diffuse lymphoma of small bowel with bleeding from only a

small segment of jejunum, injection of methylene blue intraoperatively through a previouslyplaced angiographic catheter stained the bleeding segment of jejunum blue. This segment was

identified easily and resected. These preoperative and intraoperative localisation procedures weresimple and effective and we recommend them to be used more freely.

Gastrointestinal bleeding is a common emergency,accounting for about 500 to 1200 admissions permillion population per year.'" Fortunately most ofthese bleeding lesions are situated at sites easilyaccessible to endoscopic examination and diagnosiscan speedily be made in 80 to 90% of patients byemergency oesophagogastroduodenoscopy alone.'In the minority that remains undiagnosed afterconventional investigations, many will never bleedagain.' A problem, however, arises when bleedingbecomes massive, continuous or recurrent, andwhen the bleeding lesions are malignant. The investi-gation of such patients is notoriously difficult andvery often a disproportionately large amount of

Address for correspondencc: Dr W Y Lau, Governmcnt Surgical Unit, QueenMary Hospital, Pokfulam Road, Hong Kong.

Received for publication 7 November, 1986.

medical resources and time are used."' Localisationof the bleeding source is important in the manage-ment because once it is found, diagnosis becomessimple and a rational and hopefully curative treat-ment can be offered. Localisation can be donepreoperatively or intraoperatively with a wide rangeof investigatory procedures, the correct choice ofwhich can sometimes be difficult. Our experience inthe management of such patients in the past six yearsform the basis of this report.

Methods

PATIENTSBetween 1979 and 1985, 2751 patients with gastro-intestinal bleeding were admitted into our unit.Thirty four patients (1-2%) presented with bleedingof obscure origin which is defined as a failure to find

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the source of bleeding after conventional investiga-tions. These include at least one, and often more,emergency upper gastrointestinal endoscopy, bariummeal, sigmoidoscopy, barium enema and colono-scopy. During this period, there were two patientswith Peutz-Jeghers syndrome and one with bluerubber bleb naevus syndrome."' The facial stigmataprovided the spot diagnosis. The bleeding lesionscould occur from any part of the gastrointestinaltract, however, and the problem of locating theselesions was the same as that in other cases of bleedingof obscure origin and thus they were included in thisreview.The average age of our patients was 54 years (range

2-82 years) and the median time from the firstbleeding to diagnosis was 1-9 years (range 1 month to8 years). All of them had recurrent or massivebleeding. There were 24 men and 13 women.A careful history, including a good family history

of gastrointestinal bleeding, was routinely taken.Blood dyscrasias, clotting defects and other meta-bolic disorders were excluded by laboratory investi-gations. When conventional investigations as definedabove failed to reveal the bleeding source, thepatients were investigated as follows.

PREOPERATIVE LOCALISATION9mTc pertechnetate scan for Meckel's diverticulumwas done on young patients. If the scan was negative,or for elderly patients, barium meal and followthrough was done. This was replaced by small bowelenema in the last year of the study. When thiscontrast examination was negative, or there wasactive bleeding, selective coeliac and mesentericangiography was carried out. As angiography couldnot be done for at least one week after bariumexamination because details would be obscure by thebarium in the bowel, emergency angiography shouldbe done before any barium examination when thebleeding was active. Angiography was followed by99-Tc-labelled red blood cell scan when it failed toreveal the bleeding sites. For 36 patients, the bleed-ing lesions were localised preoperatively andlaparotomy was carried out for treatment. Diagnosticlaparotomy, however, was required immediately inone patient with very active bleeding before anyinvestigation. At operation, a large, bleeding angio-sarcoma of the ileum was found.

It is important to realise that diagnosis and localisa-tion of the bleeding lesions are not the same.Sometimes the pathological nature of the bleedinglesions are known but the bleeding sites have to belocalised as in Peutz-Jeghers and blue rubber blebnaevus syndromes; or the bleeding sites have beenlocalised by emergency angiography but the patho-logical nature of the lesions remains unknown.

Unless preoperative investigation showed thelesions to lie in anatomically fixed organs, smalllesions had still to be found intraoperatively.

INTRAOPERATIVE LOCAL ISATIONMethods include (1) palpation, (2) transillumination,(3) enterotomies and eversion of the mucosa, (4)sigmoidoscopy through enterotomies, (5) colono-scopy through mouth,"' anus," or enterotomy and (6)injection of methylene blue through previouslyplaced angiographic catheters. I'When palpation and transillumination failed to

localise the lesions, the other special intraoperativeprocedures mentioned were used. These procedurescould also be used in (1) multiple small bleedinglesions in the small intestine when it was difficult tolocalise all lesions by palpation and transillumina-tion, (2) extensive tumour involvement of theintestine with bleeding only from a short segment ofintestine, (3) massive bleeding requiring emergencysurgery before preoperative localisation was possibleand (4) diagnostic laparotomy when preoperativelocalisation failed to find the lesion.

Results

PREOPERATIVE l OCAILISATION (Table 1)"'"Tc pertechnetate scan was carried out on 10patients below the age of 30. Five out of six patientswho were later confirmed to have Meckel's diverti-culi had a positive scan, giving a false negative rate of17%.Barium meal and follow through was done on 21

patients and small bowel enema on six. Eight out ofthe 27 examinations revealed either tumours orulcers in the small intestine. In a patient who hadPolya gastrectomy 10 years ago, gastroscopy andselective angiography revealed bleeding from theafferent loop. Barium meal and follow through,however, failed to show the afferent loop. A catheterwas introduced into the afferent loop through agastroscope and injection of barium revealed a lesionat the end of it. Laparotomy showed it to be aleiomyosarcoma (Fig. 1). These barium examina-tions, however, had their limitations. Only largerpolyps in the two patients with Peutz-Jeghers syn-drome were shown up. Most of the smaller polypshad to be found at operation by intraoperativeenteroscopy. In a patient with aortoduodenal fistulawith a thrombus in the duodenum, barium meal andfollow through misdiagnosed it to be a duodenaltumour (Fig. 2). In the last year of our study, sixpatients had small bowel enema; although smallbowel enemas were technically more difficult toperform than barium meal and follow through, theygave much better delineation of small bowel lesions.

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Preoperative and intraoperative localisation ofgastrointestinal bleeding ofobscure origin

Table 1 Usefulness ofpreoperative diagnostic tests in bleeding ofobscure origin*

Selective R B CPathology Patients (n) Meckel's scan Ba Meal FT/small angiography scan

bowelenema

TumoursSmooth muscle tumours of gut 6 - 4/4 2/2Lymphoma of gut 4 2/3 2/2 1/1Angiosarcoma of ileum IAfferent loop Iciomyosarcoma I - 0/1Carcinoma ofjejunum 1 - 1/1 - -

Arteriovenous malformationAVM small bowel 3 0/1 0/2 3/3 0/1A VM rectum 1 0/1 0/1 1/1Angiodysplasia of caecum 4 - 0/2 4/4

OthersMeckel's diverticuli 7 5/6 0/4 1/2 2/2Jejunal diverticuli 1 - 0/1 1/1Tuberculous ileal ulcers 1 0/1 0/1 1/1 -

Non-specific ileal ulcers 2 0/1 1/2 1/2 1/1Leaking aortoduodenal fistula 1 - 0/1Ischaemic colitis 1 - 0/1 1/1 1/1

HereditaryPeutz-Jeghers syndrome 2 - 2/2t 2/2Bluerubber blebnaevussyndrome I 1/It 1/1 -

Total 37 5/10 11/27 20/22 5/6

*Usefulness=number of diagnostic tests/number of tests carried out; tonly some lesions detected.

® ®k1 -

Fig. 1 (a) Leiomyosarcoma ofthe afferent loop; (b) tumourshown by injection ofbarium through a gastroscope.

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Fig. 2 (a) A thrombusfrom an aorto-duodenalfistula found in duodenum; (b) barium meal andfollow through showed a'tumour' in duodenum.

Coeliac and mesenteric angiography was found tobe most valuable in the diagnosis and localisation ofvascular lesions (Fig. 3). It was especially useful inpatients who were bleeding actively and in patientswho had multiple lesions in the small intestine.Emergency angiography was performed wheneverthe rate of bleeding was greater than 1 ml per minuteto give a reasonable chance to show the bleedingsites. In 11 patients, angiography was done beforebarium examinations of the small intestine. Thebleeding sites were shown up in 10 patients while99mTc-labelled red blood cell scan demonstrated thelesion in the last patient. Seven of these patientsunderwent emergency operations without furtherinvestigations because of active bleeding. For theother four patients, the bleeding stopped afterangiography and subsequent barium examinationsdiagnosed the nature of the bleeding lesions. Asangiography only revealed the bleeding sites in thesefour patients, these two tests were complementaryto each other. In a patient with multiple jejunaldiverticuli and another patient with extensive small

intestinal lymphoma shown on barium studies,angiography located the bleeding sites at theproximal jejunum (Fig. 4) and these show that boththese tests are sometimes necessary. In eight patientswho had been investigated previously for chronicand slow gastrointestinal bleeding with bariumexaminations of the small intestine without locatingthe lesions, emergency angiography done on sixpatients during acute bleeding and elective angio-graphy on two patients demonstrated the lesions. Inthree patients with multiple lesions in the gastro-intestinal tract like the Peutz-Jeghers and bluerubber bleb naevus syndromes, only the largerlesions were demonstrated by both barium studiesand angiography. The rest of the smaller lesions hadto be found intraoperatively by intraoperativelocalisation. In summary, emergency angiographygave valuable information in 18 of 20 examinationsand elective angiography done twice gave additionalinformation to other tests.

'-Tc-labelled red blood cell scan was carried outon six occasions. Five out of six examinations were

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Fig. 3 (a) Superiormesenteric angiography showed actively bleeding lesion injejunum; (b) contrast injejunum; (c) thebleeding lesion was an arteriovenous malformation.

positive. On the occasion when it failed, the bleedinghad already stopped. This scan could detect a slowerrate of bleeding than angiography but it could onlygive a rough localisation of the bleeding site.The bleeding lesions were localised preoperatively

in 36 patients. We had to do diagnostic laparotomyonly once on a patient with such profuse bleeding thatemergency operation had to be performed immedi-ately. At operation, a large bleeding angiosarcoma ofthe ileum was found.

INTRAOPERATIVE LOCALISATION (Table 2)By simple palpation and transillumination, lesionswere detected in 24 of the 37 patients though only

part of lesions were palpable in the three patientswith Peutz-Jeghers syndrome and blue rubber blebnaevus syndrome.

Multiple enterotomies and eversion of mucosa wasmessy and troublesome. It was used only once in a

patient with blue rubber bleb naevus syndrome.The haemangiomas in the small intestine were so

numerous that the whole small intestine could beinspected with this method" after their removal.

Intraoperative enteroscopy was done on 15patients. This was accomplished by a fibreopticcolonoscope which was inserted through the anus in14 patients and through an enterotomy (for excisionof polyp) and the anus in one patient. The technique

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874 Lau, Fan, Wong, Wong, Poon, Chu, Yip, and Wong

Fig. 4 (a) Multiplejejunal diverticuli; (b) emergency angiography showed bleeding lesion injejunum.

Table 2 Usefulness ofintraoperative tests in bleeding ofobscure origin*

Sigmoidoscopy Methlyletne bluePalpation+ through Fibreoptic through previously

Pathology Patients (n) Transillumination enterotomies enteroscopy placed catheter

TumoursSmooth muscle tumours of gut 6 6/6 - - -Lymphoma of gut 4 2/4 - 1/1 1/1Angiosarcoma of ileum 1 1/1 - - -Afferent loop leiomyosarcoma 1 1/1 -

Carcinoma of jejunum 1 1/1 -

Arteriovenous malformationA V M small bowel 3 0/3 - 3/3 -

A V M rectum 1 0/1 - 1/1 -

Angiodysplasia of caccum 4 0/4 - 2/4 -

OthersMeckel's diverticuli 7 7/7 - - -

Jejunal diverticuli 1 1/1Tuberculous ileal ulcers 1 1/1 -

Non-specific ileal ulcers 2 0/2 - 2/2Leaking aorto-duodenal fistula 1 1/1 -

Ischaemic colitis 1 0/1 - 1/1Hereditary

Peutz-Jeghers syndrome 2 2/2t - 2/2Blue rubber bleb naevus syndrome 1 1/it 1/1 1/1 -

Total 37 24/37 1/1 13/15 1/1

*Usefulness= number of diagnostic tests/number of tests carried out; tonly some lesions detected.

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Preoperative and intraoperative localisation ofgastrointestinal bleeding ofobscure origin

has been well described.'" This is an easy procedure,the abdominal surgeon helping to manipulatethe tip of the fibreoptic enteroscope up the colonthrough the ileocaecal valve into the terminal ileum.The whole small intestinal mucosa could be inspectedby telescoping the small bowel onto the enteroscope.This procedure detected lesions that failed to bedetected grossly in nine patients, helped to detectadditional lesions in three patients and ruled outsuspicious lesion in the jejunum shown on smallbowel enema in another patient. In two patients withcaecal angiodysplasia, it failed to detect the lesions.Preoperative tests located the bleeding sites to be inthe caecum, however, and right hemicolectomyprovided cure in both cases.On only one occasion was injection of methylene

blue through a previously placed angiographiccatheter required. In this patient who had extensivesmall bowel lymphoma with bleeding only from asmall segment of jejunum, the bleeding site wasfound by selective angiography preoperatively. Thecatheter was left in situ. At operation, injection ofmethylene blue stained the segment of jejunumcontaining the bleeding ulcers blue. This segmentwas identified easily and resected.

Discussion

Investigation of patients with bleeding of obscureorigin in the gastrointestinal tract is usually timeconsuming and difficult. The history given by patientsis rarely helpful. A strong family history with facialstigmata might suggest haemorrhagic telangiectasia,Peutz-Jeghers or blue rubber bleb naevus syndrome.Haematemesis is a reliable clue that bleeding isoccurring above the duodenojejunal flexure. Thecolour of the stool is not useful in predicting the siteof bleeding as it depends on the transit time of theblood bolus, the amount of blood as well as on theactual site.'4Emergency oesophagogastroduodenoscopy should

be a routine as acute mucosal lesions can be transientand heal rapidly.'"6 A side view duodenoscopy isuseful in diagnosing biliary and pancreatic bleeding.'0Urgent colonoscopy also yields valuable informa-tion,'7 though technically it can be difficult whenbleeding is profuse. Colonoscopy after a normaldouble contrast barium enema also give a highdiagnostic yield. 8The small bowel enema as done by duodenal

intubation and infusion of both barium and air orcarboxymethyl cellulose solution to obtain doublecontrast pictures gives superior delineation ofsmall bowel mucosal lesions when compared with astandard barium meal and follow through examina-tion.92" It was useful in the diagnosis of tumours of

the small intestine and it should be done whenpatients present with slow and continuous bleeding inorder to diagnose or to rule out neoplastic lesions. Itis complementary to angiography when bleeding ismore profuse as angiography will most of the timereveal only the bleeding sites while the nature ofthe lesions has to be demonstrated by barium exami-nations. The limitation of barium studies is thatsuperficial mucosal lesions and small vascular lesionsmay not be shown up.

Selective coeliac and mesenteric angiography hadproved to be valuable in localising bleeding sites,especially in patients with multiple gastrointestinallesions, and in diagnosis of arteriovenous malforma-tions and vascular lesions. Similar experience isshared by other authors. l 21 I Emergency angio-graphy should be carried out immediately duringactive bleeding before barium examinations asbarium in the bowel will obscure details of theangiography for at least a week.

"mTc pertechnetate scan is based on the principlethat the gamma emitting ion is secreted by thestomach and any ectopic gastric mucosa present inthe Meckel's diverticulum in the same manner aschloride, yielding a positive image with the use of agamma camera.23 Such ectopic mucosa is found in80 to 90% of patients with symptomatic Meckel'sdiverticulum.24 The false negative rate was 25 to50%25 26 and false positives are seen in a wide range ofconditions.'0 Despite these false negative and falsepositive rates, a young patient with gastrointestinalbleeding of obscure origin with a positive 9`mTcPertechnetate scan will strongly support the diag-nosis of Meckel's diverticulum.

'Tc-labelled red blood cell scan27 and OOmTcsulphur colloid scan' have the advantage that theycan detect slower rate of bleeding than angiography.In animal studies, in the former test, about 30-60 mlof blood must extravasate before it becomes positivewhile the latter test can detect blood loss of only 0 1ml/min. Unfortunately, both techniques have a falsenegative rate of 40-50% and false localisation canoccur if the isotope lost into the gut moves caudallybetween scans."' `OmTc sulphur colloid scan can berepeated every few hours, if necessary, to monitorrebleeding. In this respect it is superior to the 9 9mTc-labelled red blood cell scans which could not berepeated in a few days because of the slow decay ofthe hot spot.27 We found `mTc-labelled red blood cellscan is more sensitive than angiography in demon-strating bleeding lesions. It gives only a roughlocalisation of the bleeding sites, however, and noinformation on the nature of the bleeding lesions. Inthe era when preoperative investigations to localisethe bleeding sites were not available, laparotomyyielded a diagnosis in only about 30% of cases.9 The

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search for the bleeding site by direct examination ofthe gut is usually fruitless even if the patient has bledshortly before operation" and no lesion could befound with post mortem examination in 5-7% ofpatients.3' 32 As these patients rarely die of exsangui-nation when managed conservatively by adequateblood transfusions10 we adopted a policy of with-holding surgery as far as possible until the source ofbleeding is localised. Emergency measures of under-taking a resection (usually a partial gastrectomy orsubtotal colectomy) without having identified thebleeding site should nowadays be condemned.10With a logical plan of approach and advance in

technology, fewer and fewer patients will have tostruggle on with recurrent anaemia for months oryears or will have to take the choice of a diagnosticlaparotomy with its poor diagnostic yield. Forpatients that we fail to localise the bleeding site aftercomplete investigation, we prefer to wait. Thesepatients are closely followed up and immediatelyreinvestigated when bleeding recurs. For thosesurgeons who elect to do diagnostic laparotomy,'°intraoperative localisation should be used if grossexamination fails to find the lesions. We have alsofound intraoperative enteroscopy most valuable indetecting small lesions, especially when they aremultiple and inside the small intestine. Intraopera-tive enteroscopy is easy, effective and has littlecomplication and should therefore be used morefreely. In the rare occasion when intraoperativeenteroscopy cannot be used, as in our patient withextensive involvement of the gastrointestinal tract bylymphoma and with active bleeding from only a smallsegment of small intestine, injection of methyleneblue at operation through a previously placed angio-graphic catheter after selective angiography may behelpful in finding the bleeding lesion.

The authors wish to thank Mrs Sylvia Hulse and DrCorinna Har for their help in the preparation of thismanuscript.

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