Solitary rectal ulcer syndrome

Romanian Journal of Gastroenterology

September 2005 Vol.14 No.3, 289-291

Address for correspondence: Dimitrios K. Filippou, MD, PhD

14 Agias Eirinis Str.

GR-11146 Galatsi, Athens, Greece

Email: d_filippou@hotmail.com

Solitary Rectal Ulcer Mimicking a Malignant Stricture.

A Case Report

Evangelos Perrakis1, Antonios Vezakis1, Georgios Velimezis 1, Dimitrios Filippou2

1) Department of Surgery, Western Attica General Hospital, Athens. 2) 1st Department of General Surgery, Pireaus GP

Hospital “Tzaneio”, Greece

Abstract

Solitary rectal ulcer syndrome is a rare disorder. The

macroscopic appearance varies from single to multiple ulcers

or even circumferential lesions. A patient is presented with

solitary rectal ulcer syndrome causing circumferential

stenosis of the rectum. Solitary rectal ulcer syndrome should

be considered in all patients with malignant looking rectal

tumours. Surgical resection is usually required in patients

with rectal stricture. This can be performed either through a

laparotomy or a parasacral transsphincteric approach.

Rezumat

Ulcerul rectal solitar este o afecþiune rarã. Aspectul

macroscopic este variabil de la ulcere unice sau multiple,

pânã la leziuni circumferenþiale. Este prezentat un pacient

cu ulcer rectal solitar, care a produs o stenozã circumferenþialã

a rectului. Ulcerul rectal solitar trebuie luat în considerare la

toþi pacienþii cu tumori rectale aparent maligne. Rezecþia

chirugicalã este necesarã în prezenþa stenozei rectale.

Aceasta poate fi efectuatã fie prin laparotomie fie prin abord

transsfincterian parasacral.

Key words

Solitary rectal ulcer - surgical resection - parasacral ap-

proach

Introduction

Solitary rectal ulcer syndrome was first described in 1829

(1) but the clinicopathological features were defined by

Madigan and Morson in 1969 (2). Its incidence was estimated

at one in 100,000 per year in a 10-year study in Northern

Ireland (3). The etiology remains obscure. The macroscopic

appearance varies from erythema to ulceration or polypoid

lesions, usually on the anterior rectal wall, but sometimes it

may be more extensive, even circumferential.

A patient is discussed, with solitary rectal ulcer syndrome

causing circumferential stenosis of the rectum.

Case report

A 52 year old lady presented with altered bowel habits

(constipation) and a feeling of inadequate defecation. She

had no history of constipation in the past or excessive

straining at stools. She had been a regular user of analgesic

suppositories (paracetamol 400 mg plus codeine 20 mg plus

caffeine 50mg) for the relief of migraine during the last year.

Rectal examination revealed a hard ulcerated lesion at 3

cm on the posterior rectal wall occupying nearly half the

circumference of the rectum. The lesion was thought to be

malignant, biopsies were taken and the patient was placed

on the list for an abdominoperineal resection. There was no

evidence of visible rectal prolapse, while straining in the

squatting position. Surprisingly, the biopsies showed

chronic inflammation with fibrous obliteration of the lamina

propria without any evidence of malignancy. The patient

had an MRI scan, which showed a thickened and

oedematous rectal wall with no evidence of extramural spread,

and she was discharged.

She was re-admitted a month later for examination of the

rectum under anaesthesia. This revealed a hard ulcerated

lesion occupying the whole circumference of the rectal wall

and causing a stricture (Fig.1), which allowed only a black

Nelaton catheter CH 10 (Maersk Medical A/S) to pass

through. Biopsies were taken, and the next day the patient

had a laparoscopy and a right transverse loop colostomy.

Postoperative course was uneventful and the patient

was discharged and prescribed steroid enemas twice daily.

A month later the patient was reevaluated. The stricture

remained the same despite the use of steroids and it was

decided to proceed to resection. A sleeve resection was

performed through a transsphincteric parasacral approach,

Perrakis et al290

with an end to end anastomosis. Recovery was uneventful.

The colostomy was closed two months later. Two years after

her second operation the patient remains asymptomatic, with

no ulcer seen at endoscopy (Fig.2) and with normal

defecation. The histology of the specimen showed chronic

inflammation with replacement of the lamina propria with

fibroblasts and smooth muscle cells.

Fig.1 Endoscopic view of the stricture.

Fig.2 Endoscopic postoperative view of the rectum.

Discussion

Solitary rectal ulcer syndrome is a chronic disorder. The

mean age at presentation is the third and fourth decade with

a range from 10 to more than 80 years. There is a slight

female predominance (4). The usual presenting symptoms

are the passage of mucus and blood, constipation, tenesmus,

anorectal pain, a feeling of incomplete evacuation and

straining at defecation. In one series (4) rectal bleeding was

documented in 56%, straining in 28%, passage of mucus in

18%, constipation or diarrhoea in 55%, and 26% of patients

were asymptomatic. Our patient presented with altered bowel

habits (constipation) and a feeling of incomplete evacuation.

Solitary rectal ulcer syndrome is a rare disease, the symp-

toms are not characteristic and it is usually misdiagnosed

as inflammatory bowel disease or malignancy. Our first

clinical diagnosis was rectal cancer. Biopsies are essential

in excluding malignancy.

The histologic features of solitary rectal ulcer syndrome

are well documented. The lamina propria is replaced by fibro-

blasts, smooth muscle and collagen, with associated hyper-

trophy and disorganisation of the muscularis mucosa (2).

The lesion may appear macroscopically as erythema,

single ulcer, multiple ulcers or polypoid lesion. In one series

the reported prevalence of ulceration was 57%, of polypoid

lesions 25% and of patches of hyperaemic mucosa in 18%

of patients (3). Lesions were multiple in 30% of cases in

another series (2). Usually, they appear on the anterior rectal

wall (68 - 95% of cases) approximately 6 to 10 cm from the

anal verge (4-6). In our case the ulceration was very low at 3

cm, starting from the posterior rectal wall, where it soon

became circumferential.

The pathogenesis of solitary rectal ulcer syndrome

remains obscure. Self digitation has been suggested as a

cause (7), but the lesions are often located beyond the reach

of a finger. Prolapse induced by rectal mucosal trauma or

ischaemia has been strongly implicated as a possible cause.

The incidence of rectal prolapse (either mucosal or full rectal)

in patients with solitary rectal ulcer syndrome varies from

13 to 94% (4). A combination of rectal prolapse and

paradoxical contraction of the pelvic floor have been

suggested to cause local ischaemic injury and trauma to the

rectal mucosa, resulting in ulceration. The anterior wall

mucosa 7 to 10 cm proximal to the anal verge is often the

first and largest part of the intussusceptum, as it descends

downwards into the anal canal. This corresponds to the

usual location of ulcerations seen clinically. However,

electromyography has failed to find evidence of a hyper-

active rectal sphincter in the majority of patients (4). The

possible role of ischaemia in the pathogenesis of solitary

rectal ulcer syndrome is further supported by the association

between suppositories of ergotamine (an α-adrenoreceptor

agonist with potent vasoconstrictor action) and develop-

ment of the syndrome (9). Psychological problems expressed

as a disturbance in toileting behaviour have been suggested

as an important pathogenetic factor in some patients. The

improvement of symptoms with behavioural treatment (bio-

feedback) supports this as an important factor (10,11).

Our patient had no clinical evidence of rectal prolapse.

Trauma to the rectal mucosa by the suppositories could

have been the cause. This is supported by the location of

the ulcer to the posterior rectal wall and the low position at

3 cm, where the injury caused by suppositories is likely to

occur. Surprisingly, the ulcer had extended very quickly to

cause a circumferential stricture of the rectum. Steroid

enemas were not effective, as has been proposed by others

(3).

Surgery is advised for symptomatic solitary rectal ulcer

syndrome patients with rectal prolapse because of the poor

Solitary rectal ulcer syndrome 291

response to medical therapy. The procedure of choice in

patients with rectal prolapse is abdominal rectopexy (12). In

non prolapsing patients with intractable symptoms, local

excision, colonic resection or diverting colostomy are

required. In one study, resection and coloanal anastomosis

for benign rectal lesions in two patients with solitary rectal

ulcer syndrome had poor results, both requiring a colostomy

(13). Resection of the involved part of the rectum via a

parasacral transsphincteric approach is an alternative to low

anterior resection especially for very low rectal lesions. It

permits good access to the lower rectum and laparotomy is

avoided. In our case it was preceded by a laparoscopic right

transverse loop colostomy to avoid complete bowel

obstruction.

Conclusions

In conclusion, symptomatic solitary rectal ulcer syn-

drome should always be considered in patients with

malignant looking rectal tumours. In cases of circumferential

lesions causing stenosis of the rectum, surgical resection is

required. The parasacral approach remains an alternative to

low anterior resection for very low situated lesions.

2. Madigan MR, Morson BC. Solitary ulcer of the rectum. Gut

1969; 10: 871-881.

3. Martin CJ, Parks TG, Biggart JD. Solitary rectal ulcer syndrome

in Northern Ireland, 1971-1980. Br J Surg 1981; 68: 744-

747.

4. Tjandra JT, Fazio VW, Church JM, Lavery IC, Oakley JR,

Milsom JW. Clinical conundrum of solitary rectal ulcer. Dis

Colon Rectum 1992; 35: 227-234.

5. Pescatori M, Maria G, Mattana C, Vulpio C, Vecchio F. Clinical

picture and pelvic floor physiology in the solitary rectal ulcer

syndrome. Dis Colon Rectum 1985; 28: 862-867.

6. Levine DS. “Solitary” rectal ulcer syndrome. Gastroenterology

1987; 92: 243.

7. Thomson H, Hill D. Solitary rectal ulcer: always a self-induced

condition? Br J Surg 1980; 67: 784-785.

8. Kuijpers HC, Schreve RH, ten Cate Hoedmakers H. Diagnosis

of functional disorders of defecation causing the solitary rectal

ulcer syndrome. Dis Colon Rectum 1986; 29: 126-129.

9. Shpilberg O, Ehrenfield M, Abramowich D, Samra Y, Bat L.

Ergotamine-induced solitary rectal ulcer. Postgrad Med J 1990;

66: 483-485.

10. Binnie NR, Papachrysostomou M, Clare N, Smith AN. Solitary

rectal ulcer: the place of biofeedback and surgery in the treatment

of the syndrome. World J Surg 1992; 16: 836-40.

11. Eigenmann PA, Le Coultre C, Cox J, Dederding JP, Belli DC.

Solitary rectal ulcer: an unusual cause of rectal bleeding in

children. Eur J Pediatr 1992; 151: 658-60.

12. Sitzler PA, Kamm MA, Nicholls RJ. Surgery for solitary rectal

ulcer syndrome. Int J Colorectal Dis 1996; 16:136.

References

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