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410
M. Farmer
28.2 Symptoms (Box 28.1 )
Classically, patients with SRUS suffer with chronic constipation. They will often
spend prolonged periods on the toilet, which can be in excess of 30 min. They
describe having to strain excessively and may also need to use digital manual
evacuation.
Patients often describe passing mucus per rectum. This may be just when they
are defecating or can be passive passage of mucus with the additional problem of
underwear staining and hygiene issues.
Overt rectal bleeding is frequently associated with straining and mucus dis-
charge. This will typically be fresh, on the paper when wiping or in the bowl. The
bleeding may be painless or associated with a degree of discomfort and may also be
accompanied by lower abdominal pain.
Finally, patients with SRUS may complain of some form of prolapse. This can be
extremely upsetting and may also lead to a degree of urgency or even passive fecal
leakage.
28.3 Investigations
As seen from the varying presenting symptoms, the differential diagnosis is exten-
sive (Box 28.2 ). It is therefore not only important to confi rm a diagnosis of SRUS
but more importantly to rule out more serious conditions such as infl ammatory
bowel disease (IBD) or colorectal carcinoma:
Colonoscopy is essential: this will not only rule out malignancy or IBD but may •
also demonstrate rolled-up, thickened, corrugated rectal mucosa or an extensive
polypoid lesion (colitis cystica profunda). The classical appearances of a solitary
rectal ulcer may or may not be present. Biopsies will confi rm the presence of
fi bromuscular obliteration of the lamina propria [ 1 ] .
As the symptoms are similar to obstructed defecation syndrome (ODS), a defecating •
proctogram is advised to identify any pelvic fl oor prolapse. The prolapse is classi-
cally in the posterior compartment with evidence of intra-anal intussusception. The
Box 28.1: Symptoms of SRUS
Rectal bleeding
Mucus per rectum
Lower abdominal pain
Straining at defecation
Constipation
Digital manual evacuation
Urgency
Anorectal pain
411
28 Solitary Rectal Ulcer Syndrome and Pelvic Pain
presence of signifi cant internal prolapse is the key in many surgeons’ ethos in rec-
ommending surgical intervention.
Anorectal physiology may show a hypersensitive rectum and will demonstrate •
any problems with sphincter tone in the presence of leakage. Paradoxical pres-
sures may suggest the presence of anismus, which often accompanies SRUS and
may be a contraindication to surgery. Womack et al. demonstrated over activity
of the anal sphincter during evacuation and high voiding pressures in patients
with SRUS [
2 ] . These fi ndings have since been replicated with the additional
fi nding of paradoxical puborectalis contraction with SRUS [ 3 ] .
Endoanal ultrasound may show evidence of a thickened internal anal sphincter [ • 4 ] .
28.4 Treatment
28.4.1 Biofeedback
Traditionally, the emphasis has been on conservative management of this complicated
group of patients. The standard fi rst line of treatment is biofeedback [ 5 ] . Essentially,
this consists of a combination of formal bowel training, dietary manipulation, includ-
ing bulk laxatives, and counseling. Unfortunately, results vary and are often unsatis-
factory with patients having poor long-term results [ 6 ] . Nevertheless, a convincing
history of anismus necessitates this approach, and the treatment does no harm.
28.4.2 Drug Therapy
Topical therapy aims to heal the ulcer but by defi nition does not treat the underlying
cause of the ulcer. Short courses of steroid or 5-ASA enemas have also been used
with little effect [ 7 ] . Topical sucralfate has been prescribed with some success [ 8 ] .
Medication may be of use in treating anismus or urgency. Amitriptyline has been
used in the treatment of rectal pain associated with irritable bowel syndrome (IBS)
[ 9 ] . Amitriptyline has been shown to have some benefi ts for the anal pain of SRUS
in small doses of either 10 or 25 mg.
Box 28.2: Differential Diagnosis of Solitary Rectal Ulcer Syndrome
Prolapsing piles
Obstructed defecation syndrome (ODS)
Rectal Prolapse
Infl ammatory bowel disease
Lymphogranuloma venereum
Rectal cancer
412
M. Farmer
28.4.3 Argon Plasma Coagulation
The application of argon plasma coagulation in multiple sessions has been shown to
not only reduce bleeding associated with ulceration but also promote healing [
10 ] .
In a randomized controlled trial of 24 patients, 66% of patients healed with this
therapy compared with only 17% of patients receiving conservative therapy. Unlike
topical drug therapy, the argon may be treating more than the ulcer, possibly by
promoting scarring and resolution of any prolapse.
28.4.4 Surgery
Surgery may be indicated in patients with SRUS and convincing evidence of an
internal rectal prolapse. The process assumes the prolapse is the underlying cause
rather than anismus, resulting in a closed anorectal outlet. In recent years, new tech-
niques have developed to deal with this prolapse. These can be divided into resec-
tional procedures or fi xation procedures (see Chap. 29 ).
28.4.5 Resection
With the advent of modern surgical staplers, a number of devices have been
produced in order to resect rectal prolapse transanally. Several studies have
shown the safety and effi cacy of a stapled transanal resection procedure STARR
and the Contour Transtar
©
in treating ODS associated with internal rectal pro-
lapse. The Contour Transtar (follow on from the PPH-STARR device) was the
fi rst purpose-designed instrument to treat rectal prolapse by full-thickness tran-
sanal resection. This device allows resection under direct vision and also for
resection of larger prolapses than is possible with the PPH-STARR. If the cause
of the symptoms is deemed to be the prolapse and not the ulcer, then the use of
either STARR or Contour Transtar would be perfectly acceptable [ 11– 13 ] .
28.4.6 Fixation
Fixation of the rectum in the form of rectopexy is without problems. Traditional
abdominal rectopexy was performed via a laparotomy and involved various meth-
ods to dissect behind the rectum and fi x it to the sacrum. A large number of studies
have shown an unacceptable incidence of postoperative constipation after traditional
rectopexy, which is not ideal in patients with SRUS where constipation is thought to
be important in the etiology [ 14, 15 ] .
413
28 Solitary Rectal Ulcer Syndrome and Pelvic Pain
However, several studies have shown excellent functional results following laparo-
scopic ventral mesh rectopexy when dealing with internal and external rectal prolapse,
rectocele, enterocele, and SRUS, without inducing postoperative chronic constipation
[
16– 19 ] . When performing this procedure laparoscopically, there are additional ben-
efi ts for the patient, in particular less pain, shorter hospital stay, and better cosmesis.
Laparoscopic ventral rectopexy may therefore be considered as an option in the more
severe forms of SRUS with confi rmed prolapse and where conservative treatment has
failed. Further research is needed before fi rm conclusions can be made.
28.4.7 Personal View
I am convinced that the etiology of SRUS is part behavioral and part structural (pro-
lapse). It follows that there is a role for surgery in patients who demonstrate (either
clinically or radiologically) signifi cant prolapse. However, there is no doubt that in
order to achieve good long-term results for these patients, it is important to initiate
biofeedback, not only preoperatively but more importantly postoperatively to man-
age the behavioral aspects of SRUS. Once the prolapse has been corrected, there is
a greater likelihood of sustained benefi t following a course of biofeedback. This
may be from pelvic fl oor training or behavior modifi cation to treat potential anismus
and prevent the patient from straining.
28.5 Pelvic Pain
Chronic pelvic pain is a complex condition with interactions of both physiological
and psychological factors. The etiology can vary between male and female patients;
however, there are several causes common to both (Box 28.3 ).
Chronic pelvic pain can be divided into various pain syndromes. They essen-
tially relate to the different organ groups in the pelvis, that is, female/male genital
tracts, urethra/bladder, and anorectum. There are also neurological and musculosk-
eletal causes for pelvic pain. Finally, it should be borne in mind that in chronic
idiopathic perineal pain, as in SRUS, a prolapse may be the cause of the pain. The
underlying mechanism is obstructed defecation, leading to straining and conse-
quent chronic pain.
Box 28.3: Classifi cation of Pelvic Pain
Gynecological Endometriosis
Pelvic venous congestion
Pelvic adhesions
414
M. Farmer
28.5.1 Pelvic Pain Syndromes
28.5.1.1 Endometriosis
Endometriosis is defi ned as the presence of endometrial glands and stroma outside
the uterine cavity. It is the most common gynecological condition resulting in
chronic pelvic pain and accounting for 12–32% of women of reproductive age and
45–70% of adolescents undergoing laparoscopy for pain [ 20 ] . Endometriosis is
classically associated with cyclical pelvic pain, but it may also be noncyclical. Deep
dyspareunia is another typical presentation.
Endometriosis is classifi ed in four stages based on location and extent of disease
[ 21 ] . Imaging techniques such as ultrasound or MRI may miss peritoneal disease or
adhesions and therefore the defi nitive diagnosis of endometriosis can only be made
by laparoscopy or laparotomy [ 22 ] .
Treatment of endometriosis is dependent on the severity and site of disease
and also the patient’s desire for future fertility and includes medical and surgical
treatment. Oral contraceptives are the most common medical treatment. Other
treatments include danazol, progestins, and gonadotropin-releasing hormone
(GnRH).
Surgical treatment includes the removal of visible areas of endometriosis and
division of adhesions. More recently, laparoscopic uterosacral nerve ablation
(LUNA) has been proposed but effectiveness may be disappointing. A multicentre
randomized controlled trial of 487 women treated in 18 UK hospitals between 1998
and 2005 randomized to either LUNA or no LUNA laparoscopic surgery [ 23 ] . After
a mean follow-up of 69 months, there were no signifi cant differences between the
two groups.
Deep infi ltrating endometriosis is a particularly diffi cult group to treat. This con-
dition is discussed in detail in Chap. 32 .
Anorectal Levator ani syndrome
Proctalgia fugax
Coccydynia
Gastrointestinal Irritable bowel syndrome
Infl ammatory bowel disease
Urological Chronic prostatitis
Interstitial cystitis
Neurological Pudendal neuralgia
Musculoskeletal Pelvic fl oor myalgia
Myofascial pain
Psychological Physical/sexual abuse
415
28 Solitary Rectal Ulcer Syndrome and Pelvic Pain
28.5.2 Pelvic Congestion Syndrome
Pelvic congestion syndrome (PCS) is an important and undiagnosed cause for
chronic pelvic pain in women [
24 ] . The pain tends to be dull and aching with sharp
exacerbations often brought on by postural changes (standing for lengths of time)
and walking. Congestive dysmenorrhea, deep dyspareunia, and postcoital ache are
common fi ndings.
The etiology of PCS is the absence of functioning valves in ovarian veins, result-
ing in retrograde blood fl ow, venous dilatation, and eventually pelvic varicosities.
Noninvasive imaging techniques (ultrasound, CT, and MR venography) are useful in
diagnosing PCS and also in ruling out other causes of chronic pelvic pain [ 25, 26 ] .
Percutaneous venous embolization has been shown to offer excellent results and is
now viewed as the fi rst-line treatment option [ 27 ] .
28.5.3 Interstitial Cystitis (Painful Bladder Syndrome)
Interstitial cystitis (painful bladder syndrome) is a condition of chronic pelvic pain
associated with irritative voiding symptoms. The pain is often suprapubic and
related to bladder fi lling. Classic cystoscopic fi ndings are of Hunner’s ulcers.
Treatment may involve endoscopic ablation of Hunner’s ulcers if present [ 28 ] .
Intravesical injection of botulinum toxin A is also used to treat interstitial cystitis
[ 29 ] . Sacral nerve neuromodulation is now also well established as a treatment
option [ 30, 31 ] .
28.6 Perineal Pain Syndromes
28.6.1 Levator Ani Syndrome (Chronic Idiopathic Anal Pain)
Levator ani syndrome, or puborectalis spasm syndrome, is characterized by constant,
often dull anorectal pain, classically associated with tenderness in the region of pub-
orectalis on digital rectal examination. In accordance with the Rome 2 criteria, epi-
sodes of pain should last 20 min or longer. Other causes of pain such as fi ssure,
hemorrhoids, or SRUS should be excluded. The pathophysiology is poorly under-
stood, but spasm or overactivity of the anal sphincter may be a mechanism for pain.
The patient may incite tenderness on palpation of the puborectalis. The prevalence of
symptoms consistent with levator ani syndrome in the general population is between
6% and 7% and declines after the age of 45 years. It is more common in women.
Several treatment options have been tried, but none have proved satisfactory:
biofeedback [
32 ] , electrogalvanic stimulation [ 33 ] , digital massage of puborectalis,
sitz baths, local injection [ 34 ] , and antidepressant drugs. Botulinum toxin A has also
416
M. Farmer
been used to treat levator ani syndrome but also with no proven benefi t [ 35 ] . A rea-
sonable treatment algorithm is to use biofeedback as a fi rst-line treatment. If this
fails, low-dose amitriptyline followed by injection of the puborectalis sling with
botulinum toxin A is safe.
28.6.2 Proctalgia Fugax
Proctalgia fugax is defi ned by sudden, short-lived, severe, self-limiting bursts of
anorectal pain. The estimated prevalence ranges from 8% to 18% with men and
women equally affected. In a case study of 54 patients, de Parades showed that it
occurred at night in 35% of cases [ 36 ] . Clinical examination is characteristically
normal. Most patients will have infrequent attacks that are short lived, and all that is
required is reassurance. In more severe cases, treatment to induce relaxation of the
anal sphincters may be of benefi t. These may include botulinum toxin A [ 37 ] , dilti-
azem [ 38 ] , or salbutamol inhalation [ 39 ] .
28.6.3 Coccydynia
This is a syndrome of severe rectal, perineal, and sacrococcygeal pain characterized
by a burning discomfort radiating to the buttocks and thighs with reproduction of
the pain on manipulating the coccyx. Treatments are unsatisfactory, and in particu-
lar coccygectomy has poor results.
28.7 The Association of Chronic Idiopathic
Perineal Pain with Prolapse
Data from Oxford has suggested around 60% of patients with perineal pain have an
associated high-grade intrarectal prolapse [ 40 ] . This prolapse is not always obvious
on defecatory proctogram and may require examination under anesthetic to
identify.
Although the prolapse may be the driving force behind the development of pain,
it is argued that treatment of the pain should precede surgery to correct the prolapse.
This is because pain will not necessarily improve, and indeed may be exacerbated,
after prolapse surgery [
40 ] . Referral to a pain specialist for therapeutic techniques
detailed below may result in a more successful outcome if prolapse surgery is ever
contemplated.
417
28 Solitary Rectal Ulcer Syndrome and Pelvic Pain
28.8 Pudendal Neuralgia (Pudendal Pain Syndrome)
Pudendal neuralgia results in severe sharp or burning pain anywhere along the
course of the pudendal nerve. As a general rule, and in contrast to pain caused by an
underlying prolapse, the pain is often aggravated by sitting, particularly on soft
seats, and relieved by standing. The pain is in the perineum from the anus to the
clitoris or penis, essentially in line with the sensory and motor distribution of the
nerve. Onset may occur postoperatively especially after orthopedic hip surgery from
positioning or after repetitive cycling.
There are three common sites for impingement of the pudendal nerve during
its course in the pelvis. These are between the sacrotuberal and sacrospinous liga-
ments, within the pudendal canal and crossing the falciform process of the sacro-
tuberal ligament [
41 ] . The pain may be perceived in various pelvic areas,
depending on the site of impingement and the various anatomical variations that
can occur in the course and branches of the nerve. Central sensitization and
involvement of other organs and systems may also infl uence the distribution of
pain [ 42 ] .
MRI of the pelvis is essential to rule out other causes of pelvic pain. In classic
pudendal neuralgia, the MRI will usually be normal. Electrophysiological studies
may show diminished pudendal nerve latency, although these tests may be normal,
and diagnosis is made on clinical grounds.
The ideal management of pudendal neuralgia has yet to be defi ned. Options
include injection of local anesthetic and steroid at the site of the injury. This not
only reduces swelling and infl ammation at the area but also has sodium channel
blocking activity, reducing nerve irritability. The procedure is also useful in
diagnosis of the cause of the pain. Surgical decompression has also been advo-
cated with improved outcomes being clearly indicated the earlier the surgery is
carried out [ 43 ] .
Injection therapy and surgery are by no means the whole solution. In a retrospec-
tive study of 64 patients, Benson and Griffi s observed only 31% improvement in
symptoms with nerve injection and 60% improvement with surgical decompression
[ 41 ] . Other options include amitriptyline and antiepileptic drugs such as gabapentin
and pregabalin. Spinal neuromodulation may be an alternative with some evidence
of successful outcome [ 42 ] .
SRUS and chronic pelvic and perineal pain syndromes form a group of distress-
ing but poorly understood functional conditions affecting the rectum, anus, and pel-
vic fl oor. Establishing the correct diagnosis can be challenging and requires a
thorough history, careful clinical examination, and appropriate investigation.
Undiagnosed internal rectal prolapse may play a part in the etiology of a number of
these syndromes and should be considered during examination and investigation,
although simply correcting the prolapse may not resolve all of the symptoms.
Reassurance that there is not a serious cause for the symptoms is an important part
of the management of these patients.
418
M. Farmer
References
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Key Points
Solitary rectal ulcer syndrome
Misnomer as may be no or multiple ulcers •
Possible combined behavioral and mechanical etiology •
Characterized by:•
Chronic straining –
Mucus and bleeding –
Prolapse –
Exclude other causes •
Treat with:•
Biofeedback –
Topical therapy –
APC –
Surgery for the prolapse –
Pelvic pain
Multiple etiologies •
Some characteristic clinical features •
Involve a pain specialist early if appropriate •
Case Scenario Answers
A careful history and examination is paramount. More obvious diagnoses
such as fi ssures should be easily excluded. For those diagnoses that remain,
specifi c features should be sought from the history. When does the pain occur
and what makes it worse? Is it intermittent pain occurring at night and lasting
less than 30 min? Is the rectal examination normal? Is the pain more pro-
longed and associated with tenderness over the puborectalis? Does it hurt
when the coccyx is pressed? What is the distribution of the pain and is it better
on sitting or standing? How did the pain start? Did it follow an operation or
activity where nerve compression could have occurred? Are there features of
obstructed defecation?
If proctalgia fugax is suspected, simple reassurance may be all that is nec-
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injection. For levator ani syndrome, the patient may respond to biofeedback.
If this fails, low-dose amitriptyline followed by injection of the puborectalis
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419
28 Solitary Rectal Ulcer Syndrome and Pelvic Pain
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