Gut, 1969, 10, 871-881

Solitary ulcer of the rectumM. R. MADIGAN AND B. C. MORSON

From the Research Department, St Mark's Hospital, London

SUMMARY Solitary ulcer of the rectum is usually a disease of young adults of either sex which hasa characteristic appearance on sigmoidoscopy. Distinctive changes may also be seen in biopsiestaken from mucosa adjacent to the ulcer.The name 'solitary ulcer' is misleading because more than one ulcer may be present. Moreover,

there is a preulcerative phase which is clinically and histologically recognizable.The condition is essentially benign and may persist for many years unchanged. It has not responded

satisfactorily to medical or surgical methods of treatment.The cause of solitary ulcer is unknown. Different views on the pathogenesis are discussed.

The condition known as 'solitary ulcer' of therectum is uncommon and there are few references inthe literature. The name is misleading because theremay be more than one ulcer and there is a stage ofthe disease when no ulceration is present. Accordingto Haskell and Rovner (1965) the condition was firstdescribed by Cruveilhier (1829-42). Allen (1966),Epstein, Ascari, Ablow, Seaman, and Lattes (1966),and Wayte and Helwig (1967) have described whatappears to be a similar or related pathology underthe title 'Hamartomatous inverted polyps of therectum' and 'Colitis cystica profunda'. The expression'solitary ulcer' has been used at St Mark's Hospitalsince before World War II (Lloyd-Davies, 1969).The object of this paper is to describe the clinical

features, pathology, treatment, and behaviour of68 patients with solitary ulcer of the rectum seen atSt Mark's Hospital between 1931 and 1967. Forty-one of these were contacted and examined by one ofus (M.R.M.); 27 others who could not be tracedwere included because study of their case notesrevealed sufficient evidence to satisfy the criteriafor diagnosis.

CLINICAL ANALYSIS

TABLE IAGE AND SEX DISTRIBUTION OF SOLITARY ULCER OF THE

RECTUM

Age Group (yr)

10-19 20-29 30-39 40-49 50-59 60-69 Total

MaleFemaleTotal

459

157

22

78

15

4913

336

- 334 353 68

race, foreign travel, or smoking and drinking habits.There was no family history of the condition.

SYMPTOMS These are given in Table II. Severalpatients presented initially with constipation (four),rectal prolapse (two), difficulty in defaecation (two),sensation of incomplete evacuation (one), and in onesymptomless patient the ulceration was discoveredon routine examination.

Bleeding was in large amounts in five patients,four of whom required transfusion. In 33 out of the68 studied it was moderate in amount, meaning thepassage of not more than a cupful of blood. In theremainder of those with bleeding the amount passed

AGE AND SEX The age distribution is given inTable I. The youngest was aged 14 years and theoldest 68 years. The Table shows that solitary ulcerof the rectum is most common among young adults.The condition is equally common among men andwomen.

SOCIAL AND FAMILY HISTORY No significant cor-relation could be found with occupation, social class,

TABLE IISYMPTOMS OF PATIENTS WITH SOLITARY ULCER OF THE

RECTUM

Occurrene)

Bleeding on defaecationPassage of mucus on defaecationAnal or rectal pain and discomfortIncreased frequency of defaecationLower abdominal pain

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was slight. This may persist intermittently for years.The average time from the onset of symptoms to

seeking advice was 5 3 years, some patients waitingup to 10 years.

BOWEL HABIT This was regular in 38 patients (56 %)and irregular, meaning occasional episodes ofdiarrhoea or constipation, in 18 (26 %). The other 12patients relied on laxatives for regular bowel action.

MENTAL STATE It has been stated that patients withsolitary ulcer of the rectum are sometimes mentallyabnormal. Sixty (88 %) were judged normal. Eight(12 %) showed various degrees of mental disturbance.This was decided simply by observing behaviour andtalking to the patients.

DIGITAL EXAMINATION OF THE RECTUM The resultswere normal in 24 (35 %) patients. Induration wasmost noticeable in those with the nonulcerativestage, although it is usually slight and there were nocases of fixity of the rectal mucosa to the surround-ing structures.

SIGMOIDOSCOPY Forty-eight (70 %) had a singleulcer and 20 (30%) had more than one ulcer. Therewere eight patients with two ulcers and three withthree ulcers. The other nine had multiple small areasof scattered ulceration (Fig. 1).The site of the ulcers is given in Table III. It will be

seen that they are found with the greatest frequencyon the anterior aspect of the rectal wall. They arefrequently situated astride a valve of Houston. Thedistance of the ulcers from the anal margin variedfrom 3 to 15 cm, but most of them were found at7to 10cm.

TABLE IIISITE OF THE ULCERS

Site Position

AnteriorAntero-lateral

Lateral

PosteriorCircumferentialPostero-lateral

ScatteredTotal 100

right 7left 14right 8.5left 8.5

right 3left 1I5

Occurrenice(%,)

2621

17

155.54.5

11

about one third of all patients the ulcer had anirregular outline (Fig. 2). In another third, the ulcerwas linear in shape and measured from a fewmillimetres to 2 cm across. In most of the remainderthe ulcers were round or oval, but in a few therewere stellate and serpiginous forms.The base of a solitary ulcer has a characteristic

appearance. It is covered with a white, grey, oryellowish slough like wash leather. The ulceration isshallow, being only slightly depressed below thesurrounding intact mucosa. The flat edge is clearlymarked by a line of hyperaemia which sharplydemarcates the ulcer from adjacent intact mucousmembrane (Figs. 3 and 4). However, not all ulcershave this typical appearance. Occasionally the baseappears pink and granular through the slough. Thesurrounding mucous membrane is heaped up,nodular, or lumpy in appearance in about half thepatients (Fig. 5), or it may appear velvety, granular,or as punctuate red areas (Fig. 6). These changesextend for a few millimetres to a few centimetres fromthe ulcer and may be confluent if there is more thanone ulcer. The rest of the rectal mucosa is quitenormal except that in several patients a granular,appearance up to 17 cm, or greyish spots at theano-rectal ring like squamous metaplasia, wereobserved. In about a third of patients the mucosasurrounding the ulcer appeared oedematous, andmight bleed slightly when touched with the sig-moidoscope.

INVESTIGATIONS

Thirty-five of the 68 patients had barium enemaradiographs of which 22 were quite normal. Fivewere reported as having narrowing or irregularityof the rectal wall, while the remainder had apparentlyunrelated conditions of the colon, such as milddiverticular disease.A few patients had low haemoglobin values, but

there were no other abnormal haematologicalfindings.The Wassermann reaction was carried out on 44

patients and was positive in only one. Gonococcalcomplement-fixation tests were negative in 14 patientsand the Frei test was negative in eight. Examinationof faeces revealed no significant abnormalities.

OTHER ANORECTAL DISEASE

Most of the ulcers were about 2 cm in diameter

but they varied from a tiny spot, the size of a matchhead, to one large lesion measuring 5 x 3 cm. In

Thirty-three patients (48 %) had local conditions otherthan solitary ulcer. There were haemorrhoids in 22(33 %) and prolapse of the rectum in eleven (16 %).Three of these prolapses were complete, one showingthe solitary ulcer at its apex. The others were slight,

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FIG. 1. Multiple small scattered solitary ulcers.

FIGS. 3 and 4. The appearance ofa typical solitary ulcer.

FIG. 5. Nodular, lumpy mucosa surrounding an ulcer. FIG. 6. Punctate red mucosa surrounding an ulcer.

FIG. 7. Lumpy, polypoid, hyperaemic mucosa.

FIG. 2. Irregular ulcer.

FIG. 8. Puckei-ed scap-red-tnucosa.

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ranging from a tendency to mucosal pouting onlyto partial prolapse. Three patients had lax analsphincters with a bulging perineum. Two patientshad operations for correction of the prolapse but theulcers persisted.

Other associated conditions included leucoplakiaand excoriation of the anal skin (seven patients) andfissure-in-ano (seven patients). One patient also hadan unrelated carcinoma of the rectum.

TREATMENT

Many empirical treatments have been tried andthere is very little evidence that any of them havebeen effective. Every known kind of suppository hasbeen tried for periods of several weeks to months invarying doses. The active ingredients have includedadrenaline,antihistamines, arsenic,bismuth,decicaine,hy-drocortisone and other steroids, ichthammol,mercury, and witch hazel. Likewise, steroid retentionenemas and carbenoxalone have failed. Caustics,such as silver nitrate, heavy metals, snake venom,tannic acid, and many proprietary compounds havebeen used without success. Antibiotics and chemo-therapeutic agents have been given parenterally,locally, or by mouth in varying doses, and vitaminshave also been tried.

SURGICAL TREATMENT Six patients underwent majorprocedures. Two had a resection of part of therectum, including the ulcer, with anastomosis of theremaining rectum to the pelvic colon. In bothpatients the ulcers recurred: one as an encirclingulcer at the anastomosis which 16 years laternarrowed the rectum at this point leaving someresidual hyperaemia only; the other hada recurrenceseven months postoperatively and the ulcer persistedfor two years before resolving spontaneously toleave a scarred area.

Three cases had a proximal colostomy. In one theulcer healed in six weeks, but recurred after fourmonths and then healed nine months later; then thecolostomy was closed. Nine months after that severalsolitary ulcers were still present. A year later anend-colostomy was made and the pelvic colonresected. A year after this a typical solitary ulcerwas present at 8 to 10 cm.The second patient had rectal prolapse. A trans-

verse colostomy was made, but it did not affect theulcer. Bleeding was so severe that 10 pints of bloodwere transfused in a month. Because of this, and theprolapse, the rectum was excised, the only certainmethod of cure.The third patient had a rectal prolapse for which

she had a local amputation followed by a colostomyand repair of the prolapse from above. As the ulcer

did not heal and interference was suspected, the anuswas sutured for a time, but bleeding continued.Twelve years later she still had the colostomy and agranulomatous area at the site of the ulcer.The sixth patient had a solitary ulcer at 4.9 cm

posteriorly and a carcinoma of the rectum at 14 cmanteriorly. Sixteen days after anterior resection ofthe rectum the solitary ulcer in the remaining rectumwas healed.

MINOR PROCEDURES Several patients had the ulcercauterized with the diathermy needle, which madethe ulcer worse. However, in one patient the ulcerhad healed two months after diathermy to its base.Two patients with rectal prolapse were treated,

one by submucous sclerosing injections, after whichthe ulcer healed but recurred. The second had aThiersch wire inserted and a granulomatous areatreated hy diathermy. Subsequently several solitaryulcers developed.One patient with anal stenosis and haemorrhoids

had the anus dilated and a haemorrhoidectomy.Four years later his solitary ulcer was bigger.

ADMISSION TO HOSPITAL Five patients were admittedfor rest, general build-up, examination underanaesthesia, and biopsy of the lesion. One of theserequired 5 pints of blood in 20 days. In none of themwas the ulcer changed in any way.

NON-ULCER STAGE

There seems to be a stage of this disease which doesnot show itself as an actual ulcer but which can berecognized as a localized proctitis. Typical ulcershave been observed to heal, only to reappear later.The appearance of the nonulcerated stage is thesame as that which is seen around an ulcer. Moreover,the biopsy appearances are similar.The lumpy, polypoid, hypertrophic, hyperaemic

areas of mucosa as described above correspond insize and site to those of solitary ulcers (Fig. 7). Themucosa may appear as a puckered, scarred area(Fig. 8). These areas of mucosal change are, likesolitary ulcers, surrounded by normal rectal mucosa,sharply defined and occasionally with an oedematousappearance. Close observation of the granular typeof change has shown many tiny areas of micro-ulceration. The intense redness of some of those areashas the same appearance as the red line surroundinga typical ulcer.

DURATION AND FATE OF THE ULCERS

Leaving out 16 patients who were only seen once,the average duration of observation was just over

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Solitary ucler of the rectum

FIG. 9. Normalrectal mucosa.

eight years. Ulcers change their appearance remark-ably little over many years.The first recorded case in this series is a male

patient with a typical description of a solitary ulcerof the rectum, who today, 34 years later, has anexactly similar ulcer in the same situation.

BIOPSY DIAGNOSIS

A rectal biopsy was performed in 51 (750%) of the68 patients. Twenty-one of these made no contribu-tion to the clinical diagnosis of solitary ulcer. Theprobable reason is that biopsy is not easy underoutpatient conditions. The ulcer is flat and notreadily grasped with the biopsy forceps and bleedingoccurs if too large a bite is taken.

In 30 patients the rectal biopsy showed histo-logical appearances suggestive of solitary ulcer. Themost characteristic feature is obliteration of thelamina propria of the mucous membrane in theregion of the ulcer by fibroblasts and muscle fibres

derived from the muscularis mucosae. The fibroblastslay down collagen which is intimately mixed withthe muscle fibres and together they stream towardsthe mucosal surface between the epithelial tubules(Fig. 10). The muscularis mucosae is often thickerthan normal and its fibres are splayed and incontinuity with those in the lamina propria. Thelatter shows no significant increase in the numbers ofinflammatory cells.The epithelial component of the mucous mem-

brane showing the above changes in its laminapropria sometimes shows considerable reactivehyperplasia. There is also a tendency towardsgoblet cell depletion rather than an excess of mucusproduction. The tubules sometimes show cysticdilatation. In other cases there is erosion of thesuperficial mucosa which is covered by mucus, pus,and detached epithelial cells (Fig. 1 1). The sub-mucosal layer contains an increase in the amount ofcollagen in the neighbourhood of areas of full-thickness mucosal loss, but is otherwise normal.

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FIG. 1O. Biopsyofsolitary ulcer.Epithelial tubulesare separatedfromone another andshow reactive hyper-plasia with mucindepletion. Thelaminapropria isobliterated bysmooth muscle andcollagenfibresstreaming towardsthe mucosal surfacefrom the muscularismucosae. There isalso superficialerosion ofthemucous membrane.Haematoxylin andeosin x 100.

FIG. I 1. Biopsy ofsolitary ulcer.. Thenormal laminapropria is replacedbyfibro-musculartissue streamingtowards the surfaceof the mucosa whichshows superficialulceration and cysticdilatation on thetubiules. x 100.

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Solitary ulcer of the rectum

FIG. 12. Biopsy ofsolitary ulcer. Thearrows indicatemisplaced mucousmembrane in thesubmucosa. X 100.

Y.04

Most biopsies are taken from mucous membraneclose to the area of ulceration, but occasionally theulcer or its edge may be biopsied. This will reveal a

zone of necrotic cells covering vascular granulationtissue. Beneath this there is dense fibrosis of thesubmucosal tissues which probably accounts for thecharacteristically white appearance on sigmoido-scopy. If the edge of the ulcer is biopsied, regeneratingepithelium from the adjacent mucosa will be seen

attempting to grow over the ulcerated surface. Infive of the 51 biopsies misplacAd mucosa, includingepithelial elements and lamina propria, were seen inthe submucosa (Fig. 12). This misplaced tissue isprone to undergo cystic dilatation due to retentionof mucus giving appearances which may be confusedwith adenocarcinoma. Indeed, we have knowledgeof one young patient with solitary ulcer of therectum who was treated by abdominoperinealexcision because the biopsy appearances were falselyreported as malignant. The fibro-muscular oblitera-tion of the lamina propria mentioned above cannot

be regarded as specific for the diagnosis of 'solitaryulcer' as a similar appearance is regularly seen inprolapse of the rectum. However, we have not yetseen it under any other circumstances. This histo-logical appearance can thus be described as highlysuggestive of solitary ulcer if prolapse of the rectalmucosa has been excluded. When both conditionsare present in the same patient it can only bepresumed that the histology is in some way areflection of a common feature in the pathogenesisof two separate disorders.

PATHOLOGY OF SURGICAL SPECIMENS

Two patients in this series were treated by excisionof the rectum. Examination of the surgical specimensrevealed changes of considerable interest.

SPECIMEN 1 The patient was a man aged 28 years. Thespecimen consists of the sigmoid colon and upper rectum(Fig. 13). In the upper rectum there is a flat ulcer involving

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FIG. 13. There is aflat ulcer involvingthe entire circumference ofthe upperrectum. The mucous membrane below theulcer has a nodular appearance due tothe presence ofsubmucous cysts.

FIG. 14. A clearly defined ulcer (arrowed)is present on the anterior wall ofthe upperrectum, 7 cmfrom the distal limit ofexcision. On the posterior quadrant ofthe rectum, 3 cm from the distal limit,there is a nodule, the cut surface ofwhichshows a mucous cyst in the submucosa(see Fig. 16).

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Solitary ulcer of the rectum

FIG. 15. Section ofsolitary ulcer. Thereis formation ofdensecollagen in thesubmucosa, but theother layers ofthebowel wall are notaffected. There ismisplaced mucousmembrane (arrowed)at the margin oftheulcer. Haematoxylinand eosin x 6.

FIG. 16. Mucuscyst in the sub-mucosa oftherectum. Haema-toxylin and eosinx 6.

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the entire circumference of the bowel wall for a length of3.5 cm. The mucous membrane immediately surroundinge ulcer is flat above and nodular below, but otherwisethis appears normal.

Sections show heavy fibrosis in the submucosal tissuesat the site of the ulcer, but very little inflammatory cellinfiltration. The muscularis propria is quite normal. Thesurface of the ulcer is covered with a thin layer ofgranulation tissue. The nodular appearance of the mucousmembrane immediately below the ulcer is due to thepresence of misplaced mucosa in the submucosa at theedge of the ulcer. This has undergone cystic change.The mucous membrane of the margins of the ulcer showsthe histological changes described in the section on'Biopsy diagnosis'.

SPECIMEN 2 The patient was a woman aged 42 years.The specimen consists of the sigmoid colon and upperrectum (Fig. 14). There is a clearly defined, shallow ulcer1 cm in diameter on the anterior wall of the upper rectumabout 7 cm from the distal limit of excision. It is sur-rounded by normal mucous membrane. On the posteriorquadrant of the rectum, 3 cm from the distal limit ofexcision and about 2 cm from the ulcer, there is a nodule,about 1 cm in diameter, the cut surface of which shows amucous cyst in the submucosa covered by normal mucousmembrane.

Histological sections of the ulcer show heavy sub-mucosal fibrosis covered by a layer of granulation tissue.The muscularis propria is normal. Misplaced mucousmembrane is seen at the margins of the ulcer (Fig. 15).The nodule mentioned above, which is quite separate fromthe ulcer, is a submucosal cyst lined by columnar epi-thelium and appears to be misplaced rectal mucosa(Fig. 16). The mucous membrane of the margins of theulcer shows the histological changes described in thesection on 'Biopsy diagnosis'.

These two specimens contain what appears to be thesame pathology. The mucosal changes at the marginsof the ulcers show identical appearances as thosedescribed for the biopsy diagnosis. Both specimenscontain misplaced mucous membrane in the sub-mucosa at the edge of the ulcer, and one has it at adistance from the main lesion as well.

DISCUSSION

The recognition of solitary ulcer of the rectum isrelatively easy because of the striking sigmoidoscopicappearance. Indeed, half of the cases reported herewere diagnosed solely on the clinical history andappearance of the lesion. In the other half the diag-nosis was supported by the biopsy appearances.The nonulcerative phase is also recognizable, buthas to be distinguished from other forms of proctitis.Biopsy is useful for this purpose.The only serious complication of solitary ulcer is

massive haemorrhage and this is rare. Otherwise the

condition is essentially benign, although it maypersist for a great many years. Symptoms willsometimes regress spontaneously, but if not they areacceptable to patients who have been reassured.This study has not revealed any treatment whichaffects the course of the disease.

It is most important to distinguish the appearancesof solitary ulcer from cancer of the rectum, Crohn'sdisease, proctocolitis, and specific granulomatousconditions, such as lymphogranuloma venereum.Laboratory tests, especially biopsy, are helpful.

This study has revealed little helpful informationabout the aetiology or pathogenesis of solitary ulcer,although the following factors have been givenspecial consideration.

Self-inflicted trauma is a possible cause because20% of patients admitted rectal digitation. But it isnot easy to self-digitate very far up the rectum andthe sharply demarcated appearance of the ulcer, itsvariable position, tendency to be multiple, andstationary behaviour do not support the traumatictheory of origin. In rectal clinics many patients willadmit to aiding defaecation with the finger, spoon,or other instrument, yet solitary ulcer is a rarecondition. Cutting of the finger nails and closeobservation in hospital have failed to prove thattrauma is the principal cause in any of our patients.

Sixteen patients also had rectal prolapse. Therelationship between these two conditions is notobvious, but the fibromuscular obliteration of thelamina propria of the rectal mucosa common toboth conditions may be significant. One suggestionis that excessive straining at stool may lead to internalmucosal prolapse with repeated trauma of theprolapsed mucous membrane by a contractinghypertonic anal sphincter. But most patients withsolitary ulcer have no evidence of internal prolapse.Wayte and Helwig (1967) have described the

pathology of patients with localized submucosalcysts of the rectum under the title 'Colitis cysticaprofunda' and refer to the clinical resemblancebetween their cases and those described by Haskelland Rovner (1965) as 'solitary ulcer'. Wayte andHelwig (1967) and Epstein et al (1966) believe thatthe submucosal cysts are probably the result of aninflammatory process.

Allen (1966) used the title 'Hamartomatousinverted polyps of the rectum' to describe whatappears to be the same condition as 'solitary ulcer'and localized 'colitis cystica profunda'. Our studiesgive rather more support to the hamartomatousrather than the inflammatory theory of origin,although the use of the word 'hamartoma' isquestioned. The long history, failure to respond toanti-inflammatory treatment, occurrence in youngadults, and absence of any evidence of an acquired

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Solitary ulcer of the rectum 881

process seem to point in this general direction. Wayteand Helwig (1967) speculate whether such chroniculcers could be precursors of localized submucosalcysts, but it is just as likely, in our opinion, that thereverse is true. 'Solitary ulcer' may be primarily alocalized heterotopia or congenital duplication of therectal mucosa which undergoes cystic change due toretention of secreted mucin. If such cysts burstinto the lumen there will be secondary inflammationwith ulceration. The non-ulcerative phase observedclinically, together with the boggy, lumpy texture ofthe mucosa in the neighbourhood of ulcers, couldreally be a manifestation of a preulcerative state.

We wish to thank the consultant staff of St Mark'sHospital, particularly Mr 0. V. Lloyd-Davies, FRCS,for helpful advice and permission to study their patients.

The photographs were taken by Mr Norman Mackie.We should also like to thank Mr Lloyd Soodeen fortechnical help and Miss Margaret McIntosh for secretarialassistance. The expenses of this investigation were pro-vided by the Research Endowment Funds of St Mark'sHospital.

REFERENCES

Allen, M. S., Jr. (1966). Hamartomatous inverted polyps of the rectum.Cancer, 19, 257-265.

Cruveilhier J. (1829-42). Ulcere chronique du rectum. In AnatomfiePathologique du Corps Hunmain vol. 2, livr. 25, maladies durectum, p. 4. J. B. Bailliere, Paris.

Epstein, S. E., Ascari, W. Q., Ablow, R. C., Seaman, W. B., andLattes, R. (1966). Colitis cystica profunda. Amner. J. clin.Path., 45, 186-201.

Haskell, B., and Rovner, H. (1965). Solitary ulcer of the rectum.Dis. Colon Rect., 8, 333-336.

Lloyd-Davies, 0. V. (1969). Personal communication.Wayte, D. M., and Helwig, E. B. (1967). Colitis cystica profunda.

Ampier. J. clin. Path., 48, 159-169.

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