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CASE
75
History
The patient presented in 1985 with a long history of
a sense of blockage in the rectum that resulted in
prolonged defecation. There had been minor rectal
bleeding and mucous discharge for 1 year. There was
no awareness of any prolapse. Rectal examination
revealed a firm stricture in the mid rectum, confirmed at 8 cm by sigmoidoscopy. The lumen at this
point was approximately 13mm in diameter. There
were leukoplakia-like projections on the “rim” of
the stenosis and 5 areas of shallow ulceration at this
level. The base of the ulcers was yellow-white, firm,
and surrounded by erythematous mucosa. Above
this level to 15cm, the mucosa appeared normal.
A barium enema demonstrated the abnormality
(Figure 75.1). The radiologist stated the appearances
were not exclusive of a neoplasm.
Rectopexy for a Rectal Stricture-Ulcer
Male, 38 Years
Biopsy
Biopsy of the lesion showed distortion of the
mucosal glands, chronic inflammation, submucosal
fibrosis, and squamous epithelium. These findings
along with the sigmoidoscopic appearances were
consistent with the diagnosis of solitary rectal ulcer
syndrome (SRUS).
Operation (7.29.85)
At laparotomy, there was no apparent abnormality
of the rectum or colon. There was no palpable thickening of the rectum at the level of the stenosis.
There was a deep rectoprostatic “pouch of Douglas”
and virtual absence of normal pararectal fascia
(“lateral ligaments”). The rectum was mobilized to
the pelvic floor and anterior rectopexy performed
using a 4cm wide band of monofilament polypropylene (Ripstein operation).
Figure 75.1: The barium enema on 5/14/85
(preoperative) shows an annular irregular stenosis of the
rectum with “shouldering.” Below this level, the rectum
was in spasm.
164
Figure 75.2: Barium enema on 8/19/86 (15 months after
operation) shows a persistent area of narrowing. The
rectum below this appears normal.
Diagram 75 165
Follow-Up (1990)
Four weeks after operation, the patient reported
a significant improvement in bowel function. The
sense of obstruction was greatly diminished. Frequency of defecation was reduced, and the stools
were now of normal diameter. Rectal examination
revealed the stenosis had improved and was now
soft and supple. The clinical improvement was
maintained and, 13 months after operation, the
stenosis was almost impalpable and not apparent on
sigmoidoscopy. The ulceration had healed, leaving
some erythema and persistent leukoplakia. A
barium enema 15 months after operation showed
persistent narrowing, which was surprising in view
of the clinical and sigmoidoscopic findings (Figure
75.2). When the patient was last examined 5 years
after the operation he was asymptomatic and
without recurrence of the rectal ulceration.
Comment
The clinical, endoscopic, and pathological features
of this condition were defined by the seminal publication of Madigan and Morson in 1969.
1
The ulceration is more common in the incomplete (internal
intussusception) variety of rectal prolapse.2This
patient’s rectal ulcers were classical SRUS in appearance, although circumferential ulceration and associated stricture are unusual. The stricture is related
to the marked submucosal fibrosis that occurs in
association with the ulceration. There was no rectal
intussusception detected endoscopically (proctography was not performed). Rectopexy for SRUS has
been shown to have a low success rate when not
accompanied by complete rectal prolapse,1although
Nicholls and Simson have reported symptomatic
success in 12 out of 14 patients.3In the presence of
disabling symptoms and no alternative operation,
rectopexy in this patient was undertaken as a speculative procedure. It was considered that rectal
intussusception may have been an etiological factor
but obscured by the development of stenosis. The
response to rectopexy was successful and lasting.
The persistence of the narrowing radiologically is
interesting in view of the near-normal appearance
on sigmoidoscopic examination. In the author’s
series of 216 Ripstein rectopexies (complete pro-
lapse, 180; incomplete prolapse, 36), there was only
one other patient with SRUS and stenosis. This
patient also responded well to rectopexy. The terminology and classification of the rectal prolapse
syndrome, rectal intussusception, solitary rectal
ulcer syndrome, and proctitis cystica profunda needs
an international consensus.
CASE
76
Intersphincteric Anal Fistula with Proximal Perirectal Extension
History
In June 1977, an anal fistula was treated by operation, and, although healing occurred, the rectum
was “abnormal”. The radiological demonstration of
a rectal stricture suggested the possibility of Crohn’s
disease (Figure 76.1). The patient was referred for
further management. Examination under anaesthesia revealed marked rigidity of the anorectal junction and the rectum with a long stricture of the
rectum, most marked between 10cm and 13 cm. An
internal opening was identified on the left lateral
aspect of the dentate line with an ascending track
passing anteriorly. A focus of granulation tissue at
10cm in the midline anteriorly was identified as
a probable secondary internal opening. The rectal
mucosa was pale and edematous. Biopsies at various
levels showed nonspecific inflammation.
Male, 34 Years
Operation (12.6.77)
Peranal exploration enabled only part of the fistula
track to be laid open into the lumen. Access to the
upper limit of the track was prevented by the rectal
stricture.
Operation (2.21.78)
Laparotomy revealed an extraperitoneal mass on
the left side of the rectum but no other intraabdominal abnormality. A loop ileostomy was
performed.
Operation (6.8.78)
A posterior transsphincteric approach was used with
incision of the posterior wall of the rectum to the
12cm level. Access was still difficult due to intense
fibrosis of the rectum, and strong retraction was
required to facilitate laying open the track into
the lumen. Sutures approximated each edge of the
track to the divided wall of the rectum to ensure
hemostasis and prevent premature healing
(“marsupialization”).
Figure 76.1: The barium enema examination shows a
stricture of the lower rectum. Crohn’s disease was
considered as the likely diagnosis until the fistula openings
were identified.
166
Operation (9.12.78)
The ileostomy was closed after examination
revealed healing of the fistula was complete.
Follow-Up (1980)
Bowel function was satisfactory after 4 months.
Examination under anaesthesia (28 months after the
fistulotomy) revealed sound healing of the rectum
with resolution of the stricture.
Comment
This “upside down” fistula caused significant difficulty in diagnosis until both internal openings were
identified. The Parks classification of anal fistulae
includes the upward extension of the intersphincteric fistula,1but the high level of the track and secondary internal opening in this case was unusual.
The transsphincteric operation provided the only
access possible for complete fistulotomy. The resolution of the stricture and most of the perirectal
induration had occurred within 2 months of the
fistulotomy.
Anterior
Diagram 76 167
2
1
Posterior aspect
3
4
1 Fistula track (epithelized)
2 Perirectal fibrosis
3 Rectal muscle
4 Mucosa-submucosa
CASE
77
Necrotizing Infection After Removal of “Benign” Rectal Polyp
History
On 5.1.90, endoscopic removal of a sessile rectal
polyp (15 × 15 × 3 mm) was performed. It was situated posteriorly in the lower third of the rectum.
The histology report noted severe dysplasia (“at
least carcinoma in situ”) with no evidence of
invasive carcinoma. Within a few days, an abscess
appeared in the left ischiorectal fossa, which was
drained. A fecal fistula was apparent subsequently.
The patient was referred.
Operation (5.31.90)
Examination revealed a large area of necrosis in the
left posterior wall of the rectum with undermining
of the mucosa. This was in continuity with a large
supralevator fistula. The internal opening easily
admitted an examining digit. Laparotomy, anterograde colon irrigation, and loop ileostomy were
performed.
Female, 64 Years
Operation (7.13.92)
Abdominoperineal excision of the rectum was performed. There were no intraabdominal metastases.
Pathology
There was a raised ulcer at the site of the excised
polyp that was in continuity with a small mass (8 ×
8mm) in the left parasphincteric area. Histological
examination revealed the mass was adenocarcinoma
extending into fatty tissue. The mesenteric lymph
nodes showed no evidence of carcinoma.
Follow-Up (2004)
The patient remained well until August 1996, when
a 2cm lung metastasis in the right upper lobe (RUL)
was detected upon routine chest x-ray. A right upper
lobectomy was performed. No further recurrent
disease has been detected 11 years and 7 months
since rectal excision was performed.
Progress
The fistula healed and the ileostomy was closed
(11.12.90). Follow up examinations revealed satisfactory healing until 2 years after the polyp excision.
Examination now revealed a small, firm polypoid
lesion at the site of the previous internal opening
of the fistula. Biopsy confirmed the diagnosis of
moderately differentiated adenocarcinoma. Computerized tomography (CT) examination revealed no
evidence of metastatic disease. The anal canal wall
was thickened.
Comment
Histological review of the initial polyp removed in
1990 did not reveal evidence of invasive carcinoma,
and yet this relatively small “benign” polyp was
ultimately responsible for a rectal excision and a
right lobectomy. Necrotizing infection after polyp
excision is rare. The healing of the supralevator
fistula after a defunctioning ileostomy was encouraging, but direct spread or implantation of carcinoma cells supervened. Local re-excision was
considered to be inappropriate.
168
5.31.90
Diagram 77 169
8.28.96
7.13.92
PART
IX
Various Pathology
CASE
78
Intra-Abdominal Desmoid Tumor Unassociated with Familial Adenomatous Polyposis
History
During the first year of her life, this patient was treated
for bilateral congenital dislocation of the hips. At 26
years (1956), a caesarian section was performed, as
there was “a pelvic lump present the size of a tennis
ball.” In 1975, x-rays revealed a large soft-tissue mass
in the pelvis, causing a lytic lesion in the sacrum. This
was thought to be a chondroma. In 1977, a transabdominal biopsy was performed and the diagnosis of
desmoid tumour confirmed. On clinical examination,
a firm lobulated mass filled the lower half of the
abdomen to the level of the umbilicus. Rectal and
vaginal examination revealed a hard pelvic mass
causing marked compression of the vagina and rectum.
A computerized tomography (CT) scan demonstrated
considerable destruction of the sacrum. The tumor
size was “12 × 12 × 15 cm.” Sigmoidoscopy was not
possible. A barium enema revealed sigmoid diverticulosis. At a colorectal conference (1981), the unanimous
opinion was that the lesion was inoperable.
Female, 74 Years
Figure 78.2: CT scan demonstrates the erosion of sacrum
and sacral canal. (4.24.84).
5 Fluorouracil, and carmustine was performed in
1986. None of these treatments appeared objectively
to reduce the size of the tumor (Figures 78-1 and
78-2).
Treatment
Oral delta testolactone was commenced in 1978 and
continued for 32 months. Oral sulindac was administered for 19 months (1984–1985). Intra-aterial perfusion chemotherapy with doxorubicin, vincristine,
Figure 78.1: CT scan shows the maximum transverse and
antero-posterior dimension. (4.24.84).
172
Follow-Up (2004)
Clinical and CT examination have not demonstrated any further progression of the tumor since
1981 (Figure 78-3). On CT examination, the tumor
dimensions are 12 × 13 × 15.5cm. Small areas of
Figure 78.3: CT scan reveals no increase in size in 18
years. Calcification and areas of hypodensity are apparent.
(9.30.99).
Diagram 78 173
calcification have appeared, as have stable areas
of hypodensity interpreted as cystic or necrotic
changes. The colon and bladder are markedly displaced cranially and to the right. The stretched
dome of the bladder is at the L4/5 disc level just
above the upper surface of the tumor. A left hip
replacement was performed in 2004. The patient’s
general health is good for her 74 years.
Comment
This tumor, present for 55 years is not an example
of a familial adenomatous polyposis (FAP) related
desmoid. It is better classified as pelvic fibromatosis which occurs in young females (20–35 years)
unrelated to pregnancy. Such slow growing, locally
aggressive tumors appear to arise from muscle or
aponeurosis in the pelvis. The absence of small
bowel obstruction suggests it has not primarily
involved the bowel mesentery, so common in
desmoids complicating FAP. The tumor growth has
not progressed since 1981. This could be spontaneous regression known to occur in 7% of desmoids
2
or due to 1 of the 3 nonsurgical therapies administered. The adaptation of pelvic viscera to this
massive pelvic tumor is surprising, as there are no
significant bladder or large bowel symptoms. In
addition, the involvement of the sacral canal has
not become symptomatic. Considering the risks of
attempted resection, [St Mark’s Hospital, London,
reports an operative mortality of 8 out of 22
(36.4%)3] the nonoperative treatment of this patient
appears to have been validated.
X: Bladder
X
For a full-page image of this figure see the
appendix.