Paper
Dig Dis 1990;8:294-304
Solitary Rectal Ulcer Syndrome: Aetiology, Investigation and Management E.J. T.G.
Mackle Parks
Department of Surgery, University Floor, Belfast City Hospital, Belfast, Northern Ireland Dr. E.J. Mackle, Department of Surgery, University Floor, Belfast City Hospital, Lisburn Road, Belfast BT9 7AB (Northern Ireland)
Solitary rectal ulcer syndrome is both a distressing and embarrassing condition, which is being diagnosed more commonly; this is partially due to an increased awareness of the condition. It is a benign condition affecting the rectum, mainly in young adults. It generally pursues a chronic course characterized by rectal bleeding, passage of mucus per rectum, disordered defaecation and te-nesmus. The title of the syndrome is far from ideal and can be misleading in that the lesion is not always solitary and is not always ulcer-ative. Indeed we have found in a review of 76 cases that only 31 % had a solitary ulcer while 20% had multiple ulcers, 20% had broad-based polypoidal lesions and 29% had a patchy granular hyperaemic mucosa. To attempt to overcome this confusion in terminology Du Boulay et al. [ 1 ] proposed the title ‘mucosal prolapse syndrome’. However, in some instances prolapse is not limited to the mucosa while other patients have no prolapse at all. Cruveilheir [2] in 1842 is generally recognised as having published the first recorded description of the condition. Lloyd-Davis in the 1930s is said to have conceived the term ‘solitary rectal ulcer’. Several case reports appeared intermittently in the literature but it was only in 1969 following a review of 68 cases by Madigan and Morson [3] that the condition gained wide recognition. Aetiology and Pathogenesis Solitary rectal ulcer syndrome probably does not have a single cause and several theories have been proposed to account for the histological findings and the mucosal changes which are characteristic of the condition. Developmental The polypoidal form of the lesion was felt by Allen [4] to be hamartomatous in origin and to represent a congenital malformation of the colonic mucosal glands. Madigan and Morson [3] felt that the condition may primarily be a localized heterotopia or congenital duplication of the rectal mucosa which undergoes cystic change due to retention of Solitary Rectal Ulcer Syndrome 295
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secreted mucin. Rutter [5], however, reported that Morson no longer held these views and thus this theory is nowadays less attractive. Self-inflicted Trauma
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In view of the fact that up to 70% of patients will admit to practising digitation [6] and that frequently the ulcers occur at an accessible site, it was thought for many years that the usual cause of solitary rectal ulcer was self-inflicted trauma [3, 7, 8]. However, it is difficult to accept this theory as a major factor, in that in many instances the ulcer is at a site that is inaccessible to a patient’s finger. Furthermore, avoidance of the practice does not produce healing of the ulcers. In a group of 260 homosexuals with colorectal problems the histological features typical of solitary rectal ulcer syndrome were found in only 1 patient [9]. It therefore seems more likely that digitation is the patient’s response to the symptoms rather than the cause. Infection The association of rectal ulceration and inflammation as a result of a variety of infective conditions such as tuberculosis, gonorrhoea, lymphogranuloma, amoebiasis and syphilis etc. is well recognized. However, only normal bowel flora has ever been cultured from solitary rectal ulcers. Furthermore, the typical histological changes in the syndrome do not resemble that of any known infection. Inflammatory Bowel Disease The possibility of a pre-existing proctitis has been suggested [10]. However, normal mucin composition is found in non-specific proctitis while abnormalities of mucins with a predominance of sialomucins are associated with solitary rectal ulceration [11]. Importantly, the progression of solitary rectal ulceration to total ulcerative colitis has not been seen nor does treatment with steroids or sulphasalazine have much benefit. Drug-Induced The association of solitary rectal ulceration and the use of ergotamine tartrate has been described [12, 13]. Ergotamine-induced ulcers can be distinguished from the solitary rectal ulcer syndrome by the lack of mucosal prolapse, absence of a history of difficulty in defaecation and rapid healing after stopping the drug. Some of the reported cases have been shown to have fibromuscular obliteration of the lamina propria and this is thought to be due to mucosal ischaemia induced by the ergot preparation. Ischaemia Large vessel ischaemia is unlikely to be the cause of solitary rectal ulceration. Indeed, there has only been one reported association in the literature to date [14]. The possibility of mucosal ischaemia, however, has gained wider acceptance [15–17]. It has been proposed that the morphological changes are due to one or a combination of the following factors: (a) traction forces on the submucosal vessels; (b) obliteration of the submucosal capillaries by fibromuscular proliferation; (c) pressure necrosis of the prolapsed mucosa by the anal sphincters, and (d) high intrarectal voiding pressure combined with rectal prolapse causing venous congestion. Several mechanisms have been proposed by which the rectal mucosa can be damaged and rendered ischaemic; the morphological changes could occur as a consequence (fig. 1). 296 Mackle/Parks Over-Reactive Puborectalis The role attributed to the puborectalis varies in the different theories proposed to account for the maintenance of continence. During normal defaecation in addition to the rise in the intrabdominal pressure there is descent of the pelvic floor, widening of the anorectal angle and reflex inhibition of the puborectalis muscle. In some patients with solitary rectal ulceration, the puborectalis fails to relax and can even increase in activity [18, 19]. This failure of inhibition
causes the patients to strain in order to defaecate, which, over a prolonged period, can lead to prolapse of the anterior rectal wall and thus traumatisation by the anal sphincters. Perineal Descent Up to 80% of patients with solitary rectal ulcer syndrome can be shown to have excessive perineal descent (unpublished observations). It has been postulated that excessive perineal descent allows the anterior rectal mucosa to prolapse into the anal canal during defaecation [20]. More recently it has been suggested that it is not just the anterior rectal mucosa which prolapses, but rather the full thickness of the anterior rectal wall [21]. The exact relationship of excessive perineal descent to the pathogenesis of the syndrome is difficult to assess as we have found prolapse of the anterior wall/mucosa to be present in only 18 % of those patients with excessive perineal descent (unpublished observations). Fig. 1. a Occult rectal prolapse, b Over-reactive puborectalis. c Anterior rectal wall prolapse (associated with excessive perineal descent). Solitary Rectal Ulcer Syndrome 297
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14 1210 8 6 4 2 | 1 Male (n-25) Mean age-27 jj⅞jjjijjil Female (n-51) Mean age·37 10–20 20–30 30–40 40–50 50–60 60–70 70–80 80–90 Age at onset of symptoms Fig. 2. Age and sex in 76 cases of solitary rectal ulcer syndrome. Rectal Prolapse Devadhar [22] in 1964 was the first to propose that full thickness rectal prolapse commenced as an intussusception of the rectum. Using cineradiography Broden and Snellman [23] confirmed this theory while Theuerkauf et al. [24] showed that the lead point of the intussusception commenced approximately 6–8 cm from the anal verge. Several authors have noted an association between solitary rectal ulcer syndrome and rectal prolapse [3, 15, 25, 26]. It has been postulated that a high intrarectal voiding pressure combined with rectal prolapse is the cause of solitary rectal ulcer syndrome [17]. However, this theory does not account for those patients who have no evidence of prolapse and the question why some patients have ulcers and others do not has not been answered satisfactorily. Age and Sex In our review of 76 cases of solitary rectal ulcer syndrome only one third of patients were male (fig. 2). The mean age of onset of symptoms in men was 27 years (range 12–56) and in women 37 years (range 10–83). There was a peak incidence for males in the third decade while for females it was in the third and fourth decades reflecting the wider age distribution of the condition in women. Clinical Symptoms Rectal bleeding, the passage of mucus per rectum and tenesmus are the major clinical symptoms reported in the majority of series of solitary rectal ulcer syndrome [3, 6, 26]. 298 Mackle/Parks
Usually the bleeding is slight and is bright red in colour. Occasionally the blood is dark red and may be mixed with the stool. Rarely the bleeding is excessive and necessitates blood transfusion. Patients usually also complain of the passage of mucus per rectum, often occurring several times per day. Some patients may misinterpret this symptom as being diarrhoea but careful questioning will generally reveal that the patients mean the frequent passage of mucus in response to tenesmus. Up to half of the patients will experience some leakage of mucus, but it is rare for major faecal incontinence to occur. Pruritus ani may also be a feature and this is presumably due to the leakage of mucus. Over half of the patients may complain of difficulty in defaecation and this can be very severe; we have had a patient who spent an average of 200 min per day straining at stool. In response to the difficulty in evacuation it is perhaps not surprising that many patients resort to self-digitation to assist evacuation. Rutter and Riddell [ 15] suggested that prolapsing anterior rectal mucosa blocks the anal canal causing an obstruction and that patients self-digitate in an attempt to reduce temporarily the obstruction. However, we have found on questioning patients that the majority practise self-digitation to assist evacuation rather than to relieve an obstruction. Pain is usually not a major presenting symptom but in a few patients it can be almost disabling. The pain is described as being rectal or perineal in origin in up to half of the patients, but we have found that a similar proportion of patients also complain of pain which originates in the left iliac fossa or across the lower abdomen. Patients with excessive perineal descent describe a similar pattern of pain [27] and it is therefore possible that the pain in patients with solitary rectal ulcer syndrome is due to excessive descent of the pelvic floor stretching the pelvic nerves. Clinical Signs General examination is usually unremarkable but in up to one third of patients the left colon may be palpable and tender on abdominal examination. If patients are asked to bear down while lying in the left lateral position 60% can be shown to have excessive perineal descent with the plane of the perineum descending beyond the plane of the ischial tuberosities [28]. Rectal examination may reveal poor anal tone in half of the patients and in a third of patients the anus may be patulous [26]. On digital examination an area of induration, a thickened mucosal fold or a polypoid lesion may be palpable. A characteristic of the rectal lesion is its mobility on the underlying muscle which helps to clinically distinguish it from a neoplastic condition. Sigmoidoscopy is essential in that it enables a visual assessment to be made as well as allowing a biopsy to be taken for histolog-ical confirmation. The classic ulcer is shallow with a white sloughy base surrounded by a thin rim of erythematous mucosa which blends over a variable distance with the surrounding normal mucosa. Ulcers may be single or multiple, their shape varying from small and oval to large and serpiginous. Not all lesions have a classical appearance and in 25% the lesions may be broad-based and polypoidal while in 18% the lesions consist of a patchy granular or velvety hyperaemic mucosa [26]. Solitary Rectal Ulcer Syndrome 299
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There is a tendency for the lesions to be sited on the anterior rectal wall, but they may occur at any point on the circumference. Occasionally, if the ulcers are confluent circumferentially they may cause rectal stenosis, which can occasionally make the passage of a large-diameter rigid sigmoido-scope difficult. The distance of the lesions from the anal verge varies from 4 to 15 cm with the majority occurring at between 6 and 10 cm. Although evidence of prolapse should be
sought by requesting the patient to bear down during withdrawal of the sigmoido-scope, radiological tests such as evacuation proctography are more likely to demonstrate an anatomical abnormality. Histology A histological diagnosis of solitary rectal ulcer syndrome is made by taking biopsies not from the base of an ulcer but rather from the edge or from the granular hyperaemic mucosa. The histological criteria for diagnosis have been laid down by Madigan and Morson [3] as: (1) obliteration of the lamina propria by fibromuscular proliferation of the muscularis mucosae; (2) streaming of the fibroblasts and muscle fibres up between the crypts; (3) thickening of the muscularis mucosae, and (4) if the biopsies are deep enough the presence of misplaced mucosal glands deep to the muscularis mucosae may be demonstrated. The presence of misplaced mucosal glandular elements led to use of the term ‘localised colitis cystica profunda’, which has been considered to be a separate condition. However, Levine [29] in a review of the literature considered solitary rectal ulcer syndrome and localised colitis cystica profunda to be analogous syndromes. The glandular crypts may show distortion with branching and variation in length, usually shortening but also at times lengthening. The epithelial cells can show pseudostratification, increased mitoses and a varying amount of goblet cell depletion. Histochemical analysis of the mu-cin production by the crypts can show a loss of the normal sulphomucin to sialomucin predominance. This so-called ‘transitional mucosa’ is thought to represent an exaggerated regenerative phenomenon. Staining the mucosa to demonstrate collagen has been reported to show a diffuse increase in mucosal collagen, which can be used to facilitate the differential diagnosis of solitary rectal ulcer syndrome from other inflammatory bowel diseases [30]. None of the above features can be taken as diagnostic of solitary rectal ulcer syndrome as similar histological features have been reported on biopsies of prolapsing mucosa occurring elsewhere in the gastrointestinal tract [31]. A combination of the typical histological features together with a coexistent positive history of problems with defae-cation is highly relevant. To date there has been only one report of electron microscopy being performed on 2 cases of solitary rectal ulcer and this has added little to our knowledge of the condition in that only an increase in the number of fibroblasts was demonstrated [32]. Complications Progression of the condition is unpredictable; some authors have reported little change in the condition with time [3] while 300 Mackle/Parks
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others have noted considerable change in the appearance of the lesions [6, 16]. However, there is no constant relationship between symptoms and change in appearance, if any, in the rectal lesions [6]. The most frequently reported serious complication of the syndrome is massive blood loss requiring blood transfusion [3, 26, 33]. Asymptomatic rectal stenosis has been demonstrated on barium enema [34, 35]. Symptomatic rectal stricture, however, is rare and we have only seen 1 case in a review of 76 cases. One case of a rectal ulcer progressing to pseudo-cloaca has been reported [36]; however, this case does not seem to fit the criteria for diagnosis of solitary rectal ulcer syndrome. We have seen 1 case where the syndrome was associated with vaginal leukoplakia probably on the basis of prolapse of the anterior rectal wall/posterior vaginal wall [37].
Investigation Clinical Following a detailed history sigmoidos-copy is essential to help diagnose the condition. If a lesion is seen then the sigmoido-scope should be slowly withdrawn, while the patient bears down. This may demonstrate an intussusception or anterior rectal wall prolapse. Another clinical way of demonstrating prolapse is to request the patient to strain, while squatting over a bedpan or seated on a commode during digital examination. The amount of perineal descent may be estimated using a perineometer with the patient straining in the left lateral position [38]. Using the balloon expulsion test [39] the difficulty in defaecation described by patients would appear to be predominantly an outlet problem [40]. Fig. 3. Evacuation proctogram demonstrating recto-anal intussusception. Manometric Anorectal manometry has shed little light on the aetiology of the syndrome. Snooks et al. [28] found no significant difference in resting and squeeze anal pressures between patients and controls. Keighley and Shouler [41 ] likewise found no significant alteration in anal pressures from normal. However, they found an absence of the recto-anal inhibitory reflex in 6 of the 33 patients examined. We have been unable to confirm this latter finding in patients we have investigated. Solitary Rectal Ulcer Syndrome 301 Fig. 4. Evacuation proctogram demonstrating the presence of a rectocele and failure of the puborec-talis muscle to relax (arrowed). Electrophysiological Rutter [18] was the first to study electro-physiologically the anal sphincters of patients with solitary rectal ulcer syndrome and found that 7 out of 8 patients showed inappropriate puborectalis contraction while bearing down. Other workers have also found inappropriate puborectalis contraction but in a smaller proportion of patients [28, 41]. Using a digitally directed puden-dal nerve stimulator Snooks et al. [28] were able to show that 12 out of 20 patients had evidence of damage to innervation, while single-fibre electromyography of the external sphincter showed evidence of reinner-vation of the motor units in 15 of the 20 patients. Evacuation proctography is probably the best radiological investigation of anorectal function in that it can demonstrate abnormalities in rectal configuration during evacuation, failure of the anorectal angle to open up while straining as well as the presence of excessive perineal descent [46, 47] (⅛ 3,4). Womack et al. [48] described a technique which combined simultaneous evacuation proctography, electromyography of the external sphincter and intrarectal pressure monitoring. This latter technique is of benefit in researching the condition, but probably does not contribute much extra information that is helpful in the management of the average patient.
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Radiological Barium enema examination may show ul-ceration, polypoid lesions, stricturing or no-dularity [42, 43]. However, it is rare for a radiologist to make a correct diagnosis on radiological changes alone [44, 45]. Management The management of the syndrome has not proved to be simple, probably reflecting the multiplicity of aetiological factors involved.
302 Mackle/Parks Many patients will tolerate their symptoms if they are reassured as to the nature of their problems. Adoption of a high-fibre diet and avoidance of straining at stool can help ease symptoms [6, 26, 29, 50]. It is possible that retraining of bowel habit using biofeedback techniques might be of some benefit in treating those patients without rectal prolapse, but no long-term data on the efficacy of this method exists. Topical steroids and sulphasalazine have also been tried and have been shown to be of no significant benefit [6, 26]. Various local procedures have been tried to treat or remove the rectal mucosal prolapse; however, they do nothing to correct the underlying causative factors and are of little benefit in the long term [7, 31]. Other treatments such as diverting colostomy or even resection of the rectum have also been tried with overall disappointing results [3]. If overt or occult rectal prolapse can be shown to exist then a standard abdominal rectopexy can both relieve many of the symptoms and lead to healing of ulceration [25, 26]. For those patients with severe symptoms and without evidence of overt rectal prolapse anteroposterior rectopexy has been reported as helping 12 out of 14 patients [51]. As the majority of lesions occur on the anterior rectal wall a technique which can help impede mobility of this area would appear logical and probably anterio-posterior rectopexy should be the preferred technique for operative treatment. Incomplete rectal emptying and excessive perineal descent on evacuation proctography, however, would appear to predict that anteroposterior rectopexy is associated with a poor outcome [52]. Regrettably in many cases treatment will be far from satisfactory [53]. References Du Boulay CE, Fairbrother J, Isaacson PG: Mucosal prolapse syndrome – a unifying concept for solitary rectal ulcer syndrome and related disorders. J Clin Pathol 1983;36:1264–1268. Cruveilheir J: Maladies du rectum; in Anatomie Pathologique du Corps Humain. Paris, Baillière, 1829–1842, vol 2, book 25, p4. Madigan MR, Morson BC: Solitary ulcer of the rectum. Cancer 1966;10:871–881. Allen MS: Hamartomatous inverted polyps of the rectum. Cancer 1966;19:257–265. Rutter KRP: Solitary rectal ulcer syndrome. Proc RSocMed 1975;68:22–26. Ford MJ, Anderson JR, Gilmour HM, et al: Clinical spectrum of ‘solitary ulcer’ of the rectum. Gastroenterology 1983;84:1533–1540. Kennedy DK, Hughes ESR, Masterton JP: The natural history of benign ulcer of the rectum. Surg Gynecol Obstet 1977;144:718–720. Thomson H, Hill D: Solitary rectal ulcer: Always a self-induced condition? Br J Surg 1980;67:784–785. Sohn N, Robilotti JG: The gay bowel syndrome. Am J Gastroenterol 1977;67:478–484.
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Jalen KN, Brunt PW, Maclean N, et al: Benign solitary ulcer of the rectum – a report of 5 cases. Scand J Gastroenterol 1970;5:143–147. Ehsanullah M, Filipe MI, Gazzard B: Morphological and mucus criteria for differential diagnosis of solitary ulcer syndrome and non-specific proc-titis. J Clin Pathol 1982;35:26–30. Wormann B, Hochter W, Seib H-J, et al: Ergota-mine induced colitis. Endoscopy 1985; 17:165– 166. Eckardt VF, Kanzler G, Remmele W: Anorectal ergotism: Another cause of solitary rectal ulcers. Gastroenterology 1986;91:1123–1127.
Devroede G, Beaudry R, Haddad H, et al: Discrete ulcerations of the rectum and sigmoid. Dig Dis 1973;18:695–702. Rutter KRP, Riddell RH: The solitary ulcer syndrome of the rectum. Clin Gastroenterol 1975;4: 505–530. Franzin G, Dina R, Scarpa A, et al: The evolution of the solitary ulcer of the rectum – an endoscopic and histopathological study. Endoscopy 1982; 14: 131–134. Womack NR, Williams NS, Holmfield JHM, et al: Solitary Rectal Ulcer Syndrome 303 Anorectal function in the solitary rectal ulcer syndrome. Dis Colon Rectum 1987;30:319–323. Rutter KRP: Electromyographic changes in certain pelvic floor abnormalities. Proc R Soc Med 1974;67:53–56. Lane RH: Clinical application of anorectal physiology. Proc R Soc Med 1975;68:28–30. Parks AG, Porter NH, Hardcastle J: The syndrome of the descending perineum. Proc R Soc Med 1966;59:477–482. Bartolo DCC, Roe AM, Virgee J, et al: An analysis of the rectal morphology in obstructed defaeca-tion. Int J Colon Dis 1988;3:17–22. Devadhar DSC: New thoughts on the mechanism and treatment of rectal procidentia. J Int Coll Surg 1964;42:672–681. Broden B, Snellman B: Procidentia of the rectum studied with cineradiography: A contribution to the discussion of causative mechanism. Dis Colon Rectum 1968;11:330–347. Theuerkauf FJ, Beahrs OH, Hill JR: Rectal prolapse: Causation and surgical treatment. Ann Surg 1970;17:819–835. Schweiger M, Alexander-Williams J: Solitary-ulcer syndrome of the rectum; its association with occult rectal prolapse. Lancet 1977;i: 170–171. Martin CJ, Parks TG, Biggart JD: Solitary rectal ulcer syndrome in Northern Ireland 1971–1980. Br JSurg 1981;68:744–747. Mackle EJ, Parks TG: Clinical features in patients with excessive perineal descent. J R Coll Surg Edinb 1989;34:88–90. Snooks SJ, Nicholls RJ, Henry MM, et al: Electro-physiological and manometric assessment of the pelvic floor in the solitary rectal ulcer syndrome. Br J Surg 1985;72:131–133. Levine DS: ‘Solitary’ rectal ulcer syndrome. Are ‘solitary’ rectal ulcer syndrome and ‘localised’ colitis cystica profunda analogous syndromes caused by rectal prolapse? Gastroenterology 1987;92: 243–253. Levine DS, Surawicz CM, Ajer TN, et al: Diffuse excess mucosal collagen in rectal biopsies facilitates differential diagnosis of solitary rectal ulcer from other inflammatory bowel diseases. Dig Dis Sci 1988;33:1345–1352. Rutter KRP: Solitary ulcer syndrome of the rectum: Its relationship to mucosal prolapse; in Henry MM, Swash M (eds): Coloproctology and the Pelvic Floor. Pathophysiology and Management. London, Butterworths, 1985, pp 282–298.
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Chanvitan A, Nopanitaya W: Solitary rectal ulcer; electron microscopy study of two cases. Dis Colon Rectum 1986;29:421–425. Delancy H, Hitch WS: Solitary rectal ulcer: A cause of life-threatening haemorrhage. Surgery 1974;76:830–832.
Lewis FW, Mahoney MP, Heffernan CK: The solitary ulcer of the rectum: Radiological features. Br JRadiol 1977;50:227–228. Chapa HJ, Smith HJ, Dickinson TA: Benign (solitary) ulcer of the rectum – another cause for rectal stricture. Gastrointest Radiol 1981;6:85–88. Eisenstat TE, Herbst J: Solitary rectal ulcer progressing to pseudo-cloaca: A case report. Am Surg 1979;45:57–60. Mackle EJ, Humphreys WG, Parks TG: Vaginal leukoplakia in association with solitary rectal ulcer syndrome. Ir J Med Sci 1989; 158:92. Henry MM, Parks AG, Swash M: The pelvic floor musculature in the descending perineum syndrome. Br J Surg 1982;69:470–472. Barnes PRH, Lennard-Jones JE: Balloon expulsion from the rectum in constipation of different types. Gut 1985;26:1049–1052. Mackle EJ, Parks TG: Obstructed defaecation in the solitary rectal ulcer syndrome. Colo-proctology 1988;10:113–114. Keighley MRB, Shouler P: Clinical and manometric features of rectal ulcer syndrome. Dis Colon Rectum 1984;27:507–512. Feckzo PJ, O’Connell DJ, Riddell RH, et al: Solitary rectal ulcer syndrome: Radiologic manifestations. AJR 1980;135:499–506. Castagone D, Ranzi T, Velio P, et al: Radiologic features of the solitary rectal ulcer syndrome. Eur J Radiol 1984;4:110–113. Millward SF, Bayjoo B, Dixon MF, et al: The barium enema appearances in solitary rectal ulcer syndrome. Clin Radiol 1985;36:185–189. Levine MS, Piccolello ML, Sollenberger LC, et al: Solitary rectal ulcer syndrome: A radiologic diagnosis? Gastrointest Radiol 1986;11:187–193. Mahieu P, Pringot J, Bodart P: Defecography. 1. Description of a new procedure and results in normal patients. 2. Contribution to the diagnosis of defacation disorders. Gastrointest Radiol 1984;9: 247–251,253–261. Goei R, Baeten C, Janevski B, et al: The solitary rectal ulcer syndrome: Diagnosis with defecography. AJR 1987;149:933–936. 304 Mackle/Parks Womack NR, Williams NS, Holmfield JHM, et al: New method for the dynamic assessment of ano-rectal function in constipation. Br J Surg 1985;72: 994–998. Van den Brandt-Gradel V, Huibregtse K, Tytgat GNJ: Treatment of solitary rectal ulcer syndrome with high fibre diet and abstention of straining at defecation. Dig Dis Sci 1984;29:1005–1008. Niv Y, Bat L: Solitary rectal ulcer syndrome: Clinical, endoscopic, and histological spectrum. Am J Gastroenterol 1986;81:486–491. Nicholls RJ, Simson JNL: Anteroposterior recto-pexy in the treatment of solitary rectal ulcer syndrome without overt rectal prolapse. Br J Surg 1986;73:222–224.
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Nicholls RJ, Finlay IG, Bartram CI: Can video-proctography and anorectal physiology predict the outcome after rectopexy for the solitary rectal ulcer syndrome (SRUS)? Colo-proctology 1988; 10:112. Editorial: Solitary rectal ulcer. Lancet 1988;i:513–514.
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Dig Dis 1990;8:294-304
Solitary Rectal Ulcer Syndrome: Aetiology, Investigation and Management E.J. T.G.
Mackle Parks
Department of Surgery, University Floor, Belfast City Hospital, Belfast, Northern Ireland Dr. E.J. Mackle, Department of Surgery, University Floor, Belfast City Hospital, Lisburn Road, Belfast BT9 7AB (Northern Ireland)
Solitary rectal ulcer syndrome is both a distressing and embarrassing condition, which is being diagnosed more commonly; this is partially due to an increased awareness of the condition. It is a benign condition affecting the rectum, mainly in young adults. It generally pursues a chronic course characterized by rectal bleeding, passage of mucus per rectum, disordered defaecation and te-nesmus. The title of the syndrome is far from ideal and can be misleading in that the lesion is not always solitary and is not always ulcer-ative. Indeed we have found in a review of 76 cases that only 31 % had a solitary ulcer while 20% had multiple ulcers, 20% had broad-based polypoidal lesions and 29% had a patchy granular hyperaemic mucosa. To attempt to overcome this confusion in terminology Du Boulay et al. [ 1 ] proposed the title ‘mucosal prolapse syndrome’. However, in some instances prolapse is not limited to the mucosa while other patients have no prolapse at all. Cruveilheir [2] in 1842 is generally recognised as having published the first recorded description of the condition. Lloyd-Davis in the 1930s is said to have conceived the term ‘solitary rectal ulcer’. Several case reports appeared intermittently in the literature but it was only in 1969 following a review of 68 cases by Madigan and Morson [3] that the condition gained wide recognition. Aetiology and Pathogenesis Solitary rectal ulcer syndrome probably does not have a single cause and several theories have been proposed to account for the histological findings and the mucosal changes which are characteristic of the condition. Developmental The polypoidal form of the lesion was felt by Allen [4] to be hamartomatous in origin and to represent a congenital malformation of the colonic mucosal glands. Madigan and Morson [3] felt that the condition may primarily be a localized heterotopia or congenital duplication of the rectal mucosa which undergoes cystic change due to retention of Solitary Rectal Ulcer Syndrome 295
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secreted mucin. Rutter [5], however, reported that Morson no longer held these views and thus this theory is nowadays less attractive. Self-inflicted Trauma
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In view of the fact that up to 70% of patients will admit to practising digitation [6] and that frequently the ulcers occur at an accessible site, it was thought for many years that the usual cause of solitary rectal ulcer was self-inflicted trauma [3, 7, 8]. However, it is difficult to accept this theory as a major factor, in that in many instances the ulcer is at a site that is inaccessible to a patient’s finger. Furthermore, avoidance of the practice does not produce healing of the ulcers. In a group of 260 homosexuals with colorectal problems the histological features typical of solitary rectal ulcer syndrome were found in only 1 patient [9]. It therefore seems more likely that digitation is the patient’s response to the symptoms rather than the cause. Infection The association of rectal ulceration and inflammation as a result of a variety of infective conditions such as tuberculosis, gonorrhoea, lymphogranuloma, amoebiasis and syphilis etc. is well recognized. However, only normal bowel flora has ever been cultured from solitary rectal ulcers. Furthermore, the typical histological changes in the syndrome do not resemble that of any known infection. Inflammatory Bowel Disease The possibility of a pre-existing proctitis has been suggested [10]. However, normal mucin composition is found in non-specific proctitis while abnormalities of mucins with a predominance of sialomucins are associated with solitary rectal ulceration [11]. Importantly, the progression of solitary rectal ulceration to total ulcerative colitis has not been seen nor does treatment with steroids or sulphasalazine have much benefit. Drug-Induced The association of solitary rectal ulceration and the use of ergotamine tartrate has been described [12, 13]. Ergotamine-induced ulcers can be distinguished from the solitary rectal ulcer syndrome by the lack of mucosal prolapse, absence of a history of difficulty in defaecation and rapid healing after stopping the drug. Some of the reported cases have been shown to have fibromuscular obliteration of the lamina propria and this is thought to be due to mucosal ischaemia induced by the ergot preparation. Ischaemia Large vessel ischaemia is unlikely to be the cause of solitary rectal ulceration. Indeed, there has only been one reported association in the literature to date [14]. The possibility of mucosal ischaemia, however, has gained wider acceptance [15–17]. It has been proposed that the morphological changes are due to one or a combination of the following factors: (a) traction forces on the submucosal vessels; (b) obliteration of the submucosal capillaries by fibromuscular proliferation; (c) pressure necrosis of the prolapsed mucosa by the anal sphincters, and (d) high intrarectal voiding pressure combined with rectal prolapse causing venous congestion. Several mechanisms have been proposed by which the rectal mucosa can be damaged and rendered ischaemic; the morphological changes could occur as a consequence (fig. 1). 296 Mackle/Parks Over-Reactive Puborectalis The role attributed to the puborectalis varies in the different theories proposed to account for the maintenance of continence. During normal defaecation in addition to the rise in the intrabdominal pressure there is descent of the pelvic floor, widening of the anorectal angle and reflex inhibition of the puborectalis muscle. In some patients with solitary rectal ulceration, the puborectalis fails to relax and can even increase in activity [18, 19]. This failure of inhibition
causes the patients to strain in order to defaecate, which, over a prolonged period, can lead to prolapse of the anterior rectal wall and thus traumatisation by the anal sphincters. Perineal Descent Up to 80% of patients with solitary rectal ulcer syndrome can be shown to have excessive perineal descent (unpublished observations). It has been postulated that excessive perineal descent allows the anterior rectal mucosa to prolapse into the anal canal during defaecation [20]. More recently it has been suggested that it is not just the anterior rectal mucosa which prolapses, but rather the full thickness of the anterior rectal wall [21]. The exact relationship of excessive perineal descent to the pathogenesis of the syndrome is difficult to assess as we have found prolapse of the anterior wall/mucosa to be present in only 18 % of those patients with excessive perineal descent (unpublished observations). Fig. 1. a Occult rectal prolapse, b Over-reactive puborectalis. c Anterior rectal wall prolapse (associated with excessive perineal descent). Solitary Rectal Ulcer Syndrome 297
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14 1210 8 6 4 2 | 1 Male (n-25) Mean age-27 jj⅞jjjijjil Female (n-51) Mean age·37 10–20 20–30 30–40 40–50 50–60 60–70 70–80 80–90 Age at onset of symptoms Fig. 2. Age and sex in 76 cases of solitary rectal ulcer syndrome. Rectal Prolapse Devadhar [22] in 1964 was the first to propose that full thickness rectal prolapse commenced as an intussusception of the rectum. Using cineradiography Broden and Snellman [23] confirmed this theory while Theuerkauf et al. [24] showed that the lead point of the intussusception commenced approximately 6–8 cm from the anal verge. Several authors have noted an association between solitary rectal ulcer syndrome and rectal prolapse [3, 15, 25, 26]. It has been postulated that a high intrarectal voiding pressure combined with rectal prolapse is the cause of solitary rectal ulcer syndrome [17]. However, this theory does not account for those patients who have no evidence of prolapse and the question why some patients have ulcers and others do not has not been answered satisfactorily. Age and Sex In our review of 76 cases of solitary rectal ulcer syndrome only one third of patients were male (fig. 2). The mean age of onset of symptoms in men was 27 years (range 12–56) and in women 37 years (range 10–83). There was a peak incidence for males in the third decade while for females it was in the third and fourth decades reflecting the wider age distribution of the condition in women. Clinical Symptoms Rectal bleeding, the passage of mucus per rectum and tenesmus are the major clinical symptoms reported in the majority of series of solitary rectal ulcer syndrome [3, 6, 26]. 298 Mackle/Parks
Usually the bleeding is slight and is bright red in colour. Occasionally the blood is dark red and may be mixed with the stool. Rarely the bleeding is excessive and necessitates blood transfusion. Patients usually also complain of the passage of mucus per rectum, often occurring several times per day. Some patients may misinterpret this symptom as being diarrhoea but careful questioning will generally reveal that the patients mean the frequent passage of mucus in response to tenesmus. Up to half of the patients will experience some leakage of mucus, but it is rare for major faecal incontinence to occur. Pruritus ani may also be a feature and this is presumably due to the leakage of mucus. Over half of the patients may complain of difficulty in defaecation and this can be very severe; we have had a patient who spent an average of 200 min per day straining at stool. In response to the difficulty in evacuation it is perhaps not surprising that many patients resort to self-digitation to assist evacuation. Rutter and Riddell [ 15] suggested that prolapsing anterior rectal mucosa blocks the anal canal causing an obstruction and that patients self-digitate in an attempt to reduce temporarily the obstruction. However, we have found on questioning patients that the majority practise self-digitation to assist evacuation rather than to relieve an obstruction. Pain is usually not a major presenting symptom but in a few patients it can be almost disabling. The pain is described as being rectal or perineal in origin in up to half of the patients, but we have found that a similar proportion of patients also complain of pain which originates in the left iliac fossa or across the lower abdomen. Patients with excessive perineal descent describe a similar pattern of pain [27] and it is therefore possible that the pain in patients with solitary rectal ulcer syndrome is due to excessive descent of the pelvic floor stretching the pelvic nerves. Clinical Signs General examination is usually unremarkable but in up to one third of patients the left colon may be palpable and tender on abdominal examination. If patients are asked to bear down while lying in the left lateral position 60% can be shown to have excessive perineal descent with the plane of the perineum descending beyond the plane of the ischial tuberosities [28]. Rectal examination may reveal poor anal tone in half of the patients and in a third of patients the anus may be patulous [26]. On digital examination an area of induration, a thickened mucosal fold or a polypoid lesion may be palpable. A characteristic of the rectal lesion is its mobility on the underlying muscle which helps to clinically distinguish it from a neoplastic condition. Sigmoidoscopy is essential in that it enables a visual assessment to be made as well as allowing a biopsy to be taken for histolog-ical confirmation. The classic ulcer is shallow with a white sloughy base surrounded by a thin rim of erythematous mucosa which blends over a variable distance with the surrounding normal mucosa. Ulcers may be single or multiple, their shape varying from small and oval to large and serpiginous. Not all lesions have a classical appearance and in 25% the lesions may be broad-based and polypoidal while in 18% the lesions consist of a patchy granular or velvety hyperaemic mucosa [26]. Solitary Rectal Ulcer Syndrome 299
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There is a tendency for the lesions to be sited on the anterior rectal wall, but they may occur at any point on the circumference. Occasionally, if the ulcers are confluent circumferentially they may cause rectal stenosis, which can occasionally make the passage of a large-diameter rigid sigmoido-scope difficult. The distance of the lesions from the anal verge varies from 4 to 15 cm with the majority occurring at between 6 and 10 cm. Although evidence of prolapse should be
sought by requesting the patient to bear down during withdrawal of the sigmoido-scope, radiological tests such as evacuation proctography are more likely to demonstrate an anatomical abnormality. Histology A histological diagnosis of solitary rectal ulcer syndrome is made by taking biopsies not from the base of an ulcer but rather from the edge or from the granular hyperaemic mucosa. The histological criteria for diagnosis have been laid down by Madigan and Morson [3] as: (1) obliteration of the lamina propria by fibromuscular proliferation of the muscularis mucosae; (2) streaming of the fibroblasts and muscle fibres up between the crypts; (3) thickening of the muscularis mucosae, and (4) if the biopsies are deep enough the presence of misplaced mucosal glands deep to the muscularis mucosae may be demonstrated. The presence of misplaced mucosal glandular elements led to use of the term ‘localised colitis cystica profunda’, which has been considered to be a separate condition. However, Levine [29] in a review of the literature considered solitary rectal ulcer syndrome and localised colitis cystica profunda to be analogous syndromes. The glandular crypts may show distortion with branching and variation in length, usually shortening but also at times lengthening. The epithelial cells can show pseudostratification, increased mitoses and a varying amount of goblet cell depletion. Histochemical analysis of the mu-cin production by the crypts can show a loss of the normal sulphomucin to sialomucin predominance. This so-called ‘transitional mucosa’ is thought to represent an exaggerated regenerative phenomenon. Staining the mucosa to demonstrate collagen has been reported to show a diffuse increase in mucosal collagen, which can be used to facilitate the differential diagnosis of solitary rectal ulcer syndrome from other inflammatory bowel diseases [30]. None of the above features can be taken as diagnostic of solitary rectal ulcer syndrome as similar histological features have been reported on biopsies of prolapsing mucosa occurring elsewhere in the gastrointestinal tract [31]. A combination of the typical histological features together with a coexistent positive history of problems with defae-cation is highly relevant. To date there has been only one report of electron microscopy being performed on 2 cases of solitary rectal ulcer and this has added little to our knowledge of the condition in that only an increase in the number of fibroblasts was demonstrated [32]. Complications Progression of the condition is unpredictable; some authors have reported little change in the condition with time [3] while 300 Mackle/Parks
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others have noted considerable change in the appearance of the lesions [6, 16]. However, there is no constant relationship between symptoms and change in appearance, if any, in the rectal lesions [6]. The most frequently reported serious complication of the syndrome is massive blood loss requiring blood transfusion [3, 26, 33]. Asymptomatic rectal stenosis has been demonstrated on barium enema [34, 35]. Symptomatic rectal stricture, however, is rare and we have only seen 1 case in a review of 76 cases. One case of a rectal ulcer progressing to pseudo-cloaca has been reported [36]; however, this case does not seem to fit the criteria for diagnosis of solitary rectal ulcer syndrome. We have seen 1 case where the syndrome was associated with vaginal leukoplakia probably on the basis of prolapse of the anterior rectal wall/posterior vaginal wall [37].
Investigation Clinical Following a detailed history sigmoidos-copy is essential to help diagnose the condition. If a lesion is seen then the sigmoido-scope should be slowly withdrawn, while the patient bears down. This may demonstrate an intussusception or anterior rectal wall prolapse. Another clinical way of demonstrating prolapse is to request the patient to strain, while squatting over a bedpan or seated on a commode during digital examination. The amount of perineal descent may be estimated using a perineometer with the patient straining in the left lateral position [38]. Using the balloon expulsion test [39] the difficulty in defaecation described by patients would appear to be predominantly an outlet problem [40]. Fig. 3. Evacuation proctogram demonstrating recto-anal intussusception. Manometric Anorectal manometry has shed little light on the aetiology of the syndrome. Snooks et al. [28] found no significant difference in resting and squeeze anal pressures between patients and controls. Keighley and Shouler [41 ] likewise found no significant alteration in anal pressures from normal. However, they found an absence of the recto-anal inhibitory reflex in 6 of the 33 patients examined. We have been unable to confirm this latter finding in patients we have investigated. Solitary Rectal Ulcer Syndrome 301 Fig. 4. Evacuation proctogram demonstrating the presence of a rectocele and failure of the puborec-talis muscle to relax (arrowed). Electrophysiological Rutter [18] was the first to study electro-physiologically the anal sphincters of patients with solitary rectal ulcer syndrome and found that 7 out of 8 patients showed inappropriate puborectalis contraction while bearing down. Other workers have also found inappropriate puborectalis contraction but in a smaller proportion of patients [28, 41]. Using a digitally directed puden-dal nerve stimulator Snooks et al. [28] were able to show that 12 out of 20 patients had evidence of damage to innervation, while single-fibre electromyography of the external sphincter showed evidence of reinner-vation of the motor units in 15 of the 20 patients. Evacuation proctography is probably the best radiological investigation of anorectal function in that it can demonstrate abnormalities in rectal configuration during evacuation, failure of the anorectal angle to open up while straining as well as the presence of excessive perineal descent [46, 47] (⅛ 3,4). Womack et al. [48] described a technique which combined simultaneous evacuation proctography, electromyography of the external sphincter and intrarectal pressure monitoring. This latter technique is of benefit in researching the condition, but probably does not contribute much extra information that is helpful in the management of the average patient.
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Radiological Barium enema examination may show ul-ceration, polypoid lesions, stricturing or no-dularity [42, 43]. However, it is rare for a radiologist to make a correct diagnosis on radiological changes alone [44, 45]. Management The management of the syndrome has not proved to be simple, probably reflecting the multiplicity of aetiological factors involved.
302 Mackle/Parks Many patients will tolerate their symptoms if they are reassured as to the nature of their problems. Adoption of a high-fibre diet and avoidance of straining at stool can help ease symptoms [6, 26, 29, 50]. It is possible that retraining of bowel habit using biofeedback techniques might be of some benefit in treating those patients without rectal prolapse, but no long-term data on the efficacy of this method exists. Topical steroids and sulphasalazine have also been tried and have been shown to be of no significant benefit [6, 26]. Various local procedures have been tried to treat or remove the rectal mucosal prolapse; however, they do nothing to correct the underlying causative factors and are of little benefit in the long term [7, 31]. Other treatments such as diverting colostomy or even resection of the rectum have also been tried with overall disappointing results [3]. If overt or occult rectal prolapse can be shown to exist then a standard abdominal rectopexy can both relieve many of the symptoms and lead to healing of ulceration [25, 26]. For those patients with severe symptoms and without evidence of overt rectal prolapse anteroposterior rectopexy has been reported as helping 12 out of 14 patients [51]. As the majority of lesions occur on the anterior rectal wall a technique which can help impede mobility of this area would appear logical and probably anterio-posterior rectopexy should be the preferred technique for operative treatment. Incomplete rectal emptying and excessive perineal descent on evacuation proctography, however, would appear to predict that anteroposterior rectopexy is associated with a poor outcome [52]. Regrettably in many cases treatment will be far from satisfactory [53]. References Du Boulay CE, Fairbrother J, Isaacson PG: Mucosal prolapse syndrome – a unifying concept for solitary rectal ulcer syndrome and related disorders. J Clin Pathol 1983;36:1264–1268. Cruveilheir J: Maladies du rectum; in Anatomie Pathologique du Corps Humain. Paris, Baillière, 1829–1842, vol 2, book 25, p4. Madigan MR, Morson BC: Solitary ulcer of the rectum. Cancer 1966;10:871–881. Allen MS: Hamartomatous inverted polyps of the rectum. Cancer 1966;19:257–265. Rutter KRP: Solitary rectal ulcer syndrome. Proc RSocMed 1975;68:22–26. Ford MJ, Anderson JR, Gilmour HM, et al: Clinical spectrum of ‘solitary ulcer’ of the rectum. Gastroenterology 1983;84:1533–1540. Kennedy DK, Hughes ESR, Masterton JP: The natural history of benign ulcer of the rectum. Surg Gynecol Obstet 1977;144:718–720. Thomson H, Hill D: Solitary rectal ulcer: Always a self-induced condition? Br J Surg 1980;67:784–785. Sohn N, Robilotti JG: The gay bowel syndrome. Am J Gastroenterol 1977;67:478–484.
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Jalen KN, Brunt PW, Maclean N, et al: Benign solitary ulcer of the rectum – a report of 5 cases. Scand J Gastroenterol 1970;5:143–147. Ehsanullah M, Filipe MI, Gazzard B: Morphological and mucus criteria for differential diagnosis of solitary ulcer syndrome and non-specific proc-titis. J Clin Pathol 1982;35:26–30. Wormann B, Hochter W, Seib H-J, et al: Ergota-mine induced colitis. Endoscopy 1985; 17:165– 166. Eckardt VF, Kanzler G, Remmele W: Anorectal ergotism: Another cause of solitary rectal ulcers. Gastroenterology 1986;91:1123–1127.
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Lewis FW, Mahoney MP, Heffernan CK: The solitary ulcer of the rectum: Radiological features. Br JRadiol 1977;50:227–228. Chapa HJ, Smith HJ, Dickinson TA: Benign (solitary) ulcer of the rectum – another cause for rectal stricture. Gastrointest Radiol 1981;6:85–88. Eisenstat TE, Herbst J: Solitary rectal ulcer progressing to pseudo-cloaca: A case report. Am Surg 1979;45:57–60. Mackle EJ, Humphreys WG, Parks TG: Vaginal leukoplakia in association with solitary rectal ulcer syndrome. Ir J Med Sci 1989; 158:92. Henry MM, Parks AG, Swash M: The pelvic floor musculature in the descending perineum syndrome. Br J Surg 1982;69:470–472. Barnes PRH, Lennard-Jones JE: Balloon expulsion from the rectum in constipation of different types. Gut 1985;26:1049–1052. Mackle EJ, Parks TG: Obstructed defaecation in the solitary rectal ulcer syndrome. Colo-proctology 1988;10:113–114. Keighley MRB, Shouler P: Clinical and manometric features of rectal ulcer syndrome. Dis Colon Rectum 1984;27:507–512. Feckzo PJ, O’Connell DJ, Riddell RH, et al: Solitary rectal ulcer syndrome: Radiologic manifestations. AJR 1980;135:499–506. Castagone D, Ranzi T, Velio P, et al: Radiologic features of the solitary rectal ulcer syndrome. Eur J Radiol 1984;4:110–113. Millward SF, Bayjoo B, Dixon MF, et al: The barium enema appearances in solitary rectal ulcer syndrome. Clin Radiol 1985;36:185–189. Levine MS, Piccolello ML, Sollenberger LC, et al: Solitary rectal ulcer syndrome: A radiologic diagnosis? Gastrointest Radiol 1986;11:187–193. Mahieu P, Pringot J, Bodart P: Defecography. 1. Description of a new procedure and results in normal patients. 2. Contribution to the diagnosis of defacation disorders. Gastrointest Radiol 1984;9: 247–251,253–261. Goei R, Baeten C, Janevski B, et al: The solitary rectal ulcer syndrome: Diagnosis with defecography. AJR 1987;149:933–936. 304 Mackle/Parks Womack NR, Williams NS, Holmfield JHM, et al: New method for the dynamic assessment of ano-rectal function in constipation. Br J Surg 1985;72: 994–998. Van den Brandt-Gradel V, Huibregtse K, Tytgat GNJ: Treatment of solitary rectal ulcer syndrome with high fibre diet and abstention of straining at defecation. Dig Dis Sci 1984;29:1005–1008. Niv Y, Bat L: Solitary rectal ulcer syndrome: Clinical, endoscopic, and histological spectrum. Am J Gastroenterol 1986;81:486–491. Nicholls RJ, Simson JNL: Anteroposterior recto-pexy in the treatment of solitary rectal ulcer syndrome without overt rectal prolapse. Br J Surg 1986;73:222–224.
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