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CROHN'S DISEASE WITH CARCINOMAOF THE COLONA. DAvIs, M.B., M.R.C.P.(Edip..)

Late Medical Registrar, Royal Infirmary, Sheffield*

J. P. CALEY, M.B., M.R.C.P.Senior Medical Registrar Royal Infirmary, Sheffield

Crohn's disease is not usually regarded as apremalignant state. Recent reviews of the litera-ture1' 2 make scant reference to an association ofregional enteritis with carcinoma. However, atthe World Congress of Gastro-enterology, I958,Crohn observed that he had seen two cases ofcarcinoma of the small intestine co-existing withregional enteritis.3

Rare cases of carcinoma arising in patients withpre-existing Crohn's disease have been describedin both the small and large intestine. Van Patter7mentions a case of malignant change occurring inassociation with regional enteritis of the colon,whilst Warren and Sommers8 reported an adeno-carcinoma developing in an area of Crohn'sdisease in the ascending colon. In addition,Ginzburg5 and Kornfeld6 each describe a case ofcarcinoma arising in the jejunum in areas affectedby regional enteritis. In view of this uncommon-association the following case is reported.

Case ReportM.E.S., a housewife, aged 44 years, was referred

to the surgical out-patient department in AprilI956, with a three months' history of attacks ofcolicky lower abdominal pain. The pain com-menced in the epigastrium and radiated diffuselyover the lower abdomen, tending to be worse afterfood, and being eased occasionally by alkalis.Nausea was present in the attack, but there wasno vomiting. Bowel movements were regular,except during an attack, when constipation wasnoted. Examination revealed central epigastrictenderness, but no other significant abnormality.Rectal examination was negative. A barium meal,cholecystogram, and catheter specimen of urinewere normal. Observation was undertaken in theout-patient clinic, and in July 1956 a furtherbarium meal was again reported as normal.Follow-through was not, performed. In August

*Present address; Medical Research Laboratory, P.O. Box30141, Nairobi, Kenya.

1956 the attacks of abdominal pain became in-creasingly severe, and were associated with vomit-ing of undigested food. It was therefore decidedto advise laparotomy.At operation (Mr. P. D. Livingstone) the caecum

and appendix were reddened and oedematous.The serosa of the terminal three feet of ileumshowed increased vascularity, and the mesenterywas thickened and contained large, flesh-colouredglands. No abnormality was detected in anyother viscera. An operative diagnosis of Crohn'sdisease was made, and a mesenteric gland -removedfor section. The post-operative course was un-eventful. The operative diagnosis was sub-stantiated by the histological appearances whichshowed giant cell-epithelioid cell systems withoutcentral caseation present near the periphery gland.The appearances were entirely consistent withCrohn's disease (Fig. i).

Following discharge on a low residue diet, thepatient remained well for the next six months,apart from occasional attacks of mild lower abdo-minal discomfort, and in April 1957 a bariumenema showed narrowing of the caecum with-proximal small bowel dilatation. Fifteen monthslater, in July I958, her symptoms altered. Attacksof diffuse lower abdominal pain occurred togetherwith diarrhoea, and the passage of four to sixloose stools per day containing blood and mucus.There was associated anorexia and weight loss.Over the course of the next two months hersymptoms- became increasingly severe, and inOctober I958 she was admitted to the RoyalInfirmary, Sheffield.

Examination revealed a thin, -apprehensive,afebrile woman. The abdomen was slightly dis-tended in the lower half, and there was markedtenderness in the left iliac fossa. There was nohepatosplenomegaly, nor were the kidneys pal-pable. Peristalsis was not seen. No free fluidwas detected. A healed right paramedian scarand a sebaceous cyst in the epigastrium were

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Junze I960 DAVIS and CALEY: Crohn's Disease with Carcinoma of the Colon 38I

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FIG. I

noted. The rectum was empty. Sigmoidoscopyto i i cm. was normal, but at that level a black,lustreless stool was seen, with specks of blood onits surface. There were no other significantabnormalities on physical examination. Bloodpressure 140/85 mm.Hg.

InvestigationsW.R. negative. E.S.R. (Wintrobe) 3I mm. in

one hour. Hb. I0 g./Ioo ml. (7I%) W.B.C.6,ooo.

Stool: Benzidine reaction for occult bloodstrongly positive. No ova or parasites seen.Culture for M. tuberculosis negative.

Catheter specimen of urine showed no abnor-mality.

Blood urea 2I mg./Ioo ml., serum sodiumI4I nEq./I., chloride ioo mEq./l., potassium3.7 mEq./l.Serum proteins 7.7 g./I00 ml. Electrophoresis

showed a normal pattern.A five-day fat balance study on a 5o-g. daily

fat intake showed an average daily output of2.5 g. of fat.

Chest X-ray was normal.A barium meal and follow-through showed

narrowing of the terminal ileum and caecum(Fig. 2).

Barium enema disclosed an almost complete

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FIG. 2

hold up of barium by an irregular area of narrow-ing in the sigmoid colon (Fig. 3). The radio-logical appearances were suggestive of carcinoma,but Crohn's disease of the colon could not beexcluded with certainty. In view of the radio-logical findings it was decided that a furtherlaparotomy was indicated.

Operative FindingsFollowing preparation for surgery, exploration

was undertaken on November 4, I958, by Mr.J. E. Oliver, through a left paramedian incision.A carcinoma of the pelvi-rectal junction wasfound, and conservative resection with an end-to-end anastomosis performed. There were nometastases present. In addition, the caecum andterminal ileum were found to be thickened anddensely bound down by matted adhesions pre-senting a picture of quiescent Crohn's disease.The pathological specimen comprised 28 cm.

of colon, containing a large ulcerating growth,8 cm. by 4 cm., which had invaded through thebowel wall into the adjacent fat. Histologyshowed a well-differentiated adenocarcinoma in-vading all coats of the intestine (Fig. 4). Therewas a secondary deposit in the peri-colic fat.The patient made a good recovery from opera-tion, and when last seen in February 1959 had

B1

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382 POSTGRADUATE MEDICAL JOURNAL June I960

. .Ue

FIG. 3

gained over one stone in weight and was symp-tom free.

DiscussionAlthough Crohn's disease may occur at any

age, it commonly arises in the young.3 Con-versely, the incidence of carcinoma of the colonrises with advancing years, becoming maximal inthe age groups SO to 70 years.2 The naturalhistory of Crohn's disease extends over manyyears, and the majority of patients with this con-dition will live to an age overlapping those agegroups in which colonic carcinoma reaches itspeak incidence. In view of this, one would expectoccasional association of the two conditions, butin fact the co-existence of small intestinal Crohn'sdisease and carcinoma of the colon is distinctlyunusual.The rare development of small intestinal carci-

noma on an area of regional ileitis or jejunitiscould conceivably be related to the presence of apre-existing chronic inflammatory lesion, inparallel with the occurrence of cancer arising onsites of chronic irritation elsewhere in the body.Similarly, carcinoma of the colon arising on, anarea of regional colitis is most uncommon,although the same argument is applicable.

It is interesting to note that in a recent ex-haustive study of Crohn's disease, Van Patter7

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FIG. 4

observed an oedematous, hyperaemic mucosa inthe rectum and sigmoid in a small number ofcases of Crohn's disease with no demonstrablecolonic involvement. It was considered thatthese lesions were of no particular significance.However, in two of the cases the mucosa showeda polypoid appearance. In addition, mucosalulcerations in the rectum and sigmoid were seenin 56 of 6oo cases, and when these cases wereobserved over the course of many years theyshowed a general trend to stricture formation atthe site of these ulcers. Of these 56 cases, approxi-mately one-third showed radiological evidence ofpurely small intestinal or proximal colonic Crohn'sdisease. Thus it may be accepted that, in a verysmall number of patients with small intestinal orproximal colonic Crohn's disease, non-specificmucosal changes occur in the rectum and sigmoid,in the absence of demonstrable direct colonicinvolvement. Furthermore, these changes consistof hyperaemia, oedema, or rarely a polypoidappearance, and may over the course of yearslead to a stricture in the distal colon. It is interest-ing to speculate whether carcinomatous meta-plasia of the cells of these areas of rectal andsigmoid mucosa could occur over the years, andif this mechanism could provide an explanation ofthe occurrence of malignancy in the present case.If this is so, it is surprising that the association

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Yune I960 DAVIS and CALEY: Crohn's Disease with Carcinoma of the Colon 383

of Crohn's disease and cancer of the colon is arare one, and it would seem probable that theassociation of these two conditions in this patientis fortuitous.

Recurrent abdominal symptoms in Crohn'sdisease are not uncommon--exacerbation of theoriginal disorder, subacute or acute intestinalobstruction, fistulae formation, extension of thedisease in the small gut, or extension to the largebowel usually provide the explanation. Rarely,however, malignant change may occur, and forthese reasons a full investigation of all recurrentsymptoms in Crohn's disease is essential. In thiscontext both barium meal with follow-throughand barium enema must be performed to reachan accurate diagnosis, and the importance of thelatter in the diagnosis of recurrence was stressedby Garlock and Crohn.4 Biopsy of any abnormalareas in the rectum and sigmoid should be under-taken and the attribution of recurrent abdominalsymptoms in a patient with Crohn's disease toan exacerbation of this disorder should only bemade with a full realization of the other variousdiagnostic possibilities.

SummaryA case of carcinoma of the distal colon arising

in a patient with small intestinal and proximalcolonic Crohn's disease is described. No evidenceof distal colonic Crohn's disease was found. Thedifficulties of diagnosis are noted.

AcknowledgmentsWe thank Dr. H. P. Brody and Mr. Clifford

Jones for permission to publish this case, ProfessorA. W. Kay for helpful criticism, and Dr. 0. G.Dodge for the histological reports.

REFERENCES

I. ARMI'1'AGE, (;., and WILSON, M. (1950), Brit. Y. Surg.,38, 182.

2. BOCKUS, H. L. (I944), C;astro-enterology,' vol. II, p. 755.W. B. Saunders & Co.

3. CROHN, B. B., and YARNIS, H. (1958), 'Regional Ileitis,'2nd ed. New York: Grune and Stratton.

4. GARLOCK, J. H., and CROHN, B. 13. (1945), Y. Amer. med.Ass., 127, 205.

5. GINZBLR(,, L., SCHNEIDER, K. M., DREIZIN, D. H.,and LEVINSON, C. (i956), Suirgery, 39, 347.

6. KORNFELD, P., GINZBURG, L., and ALDERSBERG, D.(ig97),-Amer. Y. Med., 23, 493.

7. VAN PATTER, W. N., BARGEN, J. A., DOCKERTY,M. B., FELDMAN, W. H., MAYO, C. W., and WAUGH,J. M. (1954), Gastroenterology, 26, 347.

8. WARREN, S., and SOMMERS, S. C. (1948), Amer. .Y. Path.,24, 475-

9. World Congress of Gastro-enterology (1958), Vol. II, p. 1342.Baltimore: The Williams & Wilkins Co.

DIETARY FAT, CHOLESTEROL METABOLISM & CORONARY DISEASE(Postgraduate Medical Journal, April 1959)

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THE EPIDEMIOLOGY OF ISCHAEMIC THE METABOLISM OF CHOLESTEROLHEART DISEASE B. Lewis, Ph.D., M.D. (Cape Town)B. Bronte-Stewart, MI.D., MI.R.C.P. PRESENT CLINICAL APPLICATIONS OF

THE REGULATION OF THE HUMAN DIET TO THE PREVENTION OFSERUM-CHOLESTEROL LEVEL ISCHAEMIC HEART DISEASEH. Gordon, M.D. (Cape Town) J. F. Brock, D.M., F.R.C.P.

THE RELATIONSHIP BETWEEN THE MYOCARDIAL INFARCTION-THE COM-SERUM LIPIDS AND THE DEVELOP- PARATIVE RACIAL PREVALENCE INMENT OF ISCHAEMIC HEART DISEASE CAPE TOWN, 1957-AN ELECTROCAR-B. Bronte-Stewart, M.D., M.R.C.P. DIOGRAPHIC STUDY

THE COAGULABILITY OF BLOOD IN V. Schrire, M.Sc., Ph.D., M.B., Ch.B. (CapeRELATION TO CORONARY HEART Town), M.R.C.P. (London and Edinburgh)DISEASE ISCHAEMIC HEART DISEASE IN AFRICANC. Merskey, M.D. (Cape), M.R.C.P. (London), POPULATIONS

and H. Lackner, M.D. (Leeds), M.R.C.P. J. F. Brock, D.M., F.R.C.P., and H. Gordon,(London) M.D. (Cape Town)

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