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Springer-Verlag 1998
O R I G I N A L A RT I C L E
Abstract Sixty-seven patients with chronic pilonidal sinuses were treated by excision and rhomboid flap transposition (RFT). Primary healing was obtained in all patients
except two who developed a seroma and one who had a
partial dehiscence of the surgical wound due to a hematoma, which necessitated drainage through the margin of
the flap. The average stay was 5.3 days (range 116). All
patients returned to normal activities within 2 weeks of surgery. No late recurrence occurred after a mean follow-up
of 74.4 months (range 8137).
Rsum 67 patients porteurs dun sinus pilonidal chronique ont t traits par excision et lambeau cutan rhombode. Une gurison premire a t obtenue chez tous les
patients lexception de deux qui ont dvelopp un srum
et un qui a dvelopp une dhiscence partielle de la plaie
en raison dun hmatome ayant ncessit un drainage par
la berge du lambeau. La dure moyenne de sjour est de
5.3 jours (116). Tous les patients ont repris une activit
normale dans les deux semaines qui ont suivi la chirurgie.
Aucune rcidive tardive na t observe au cours dun follow-up moyen de 74.4 mois (8137).
From April 1986 to April 1996, 67 patients with pilonidal sinus were
treated by excision and rhomboid flap transposition (RFT). Sixtyone (91%) were male of ages ranging from 1833 years (mean:
24 years). Fifty-one (76%) patients had one opening within the natal cleft, ten (15%) had two or three openings, always along the midline, while six (9%) also had one or more laterally situated granulation-lined openings. Six (9%) had previously been operated by excision with primary skin closure and had experienced recurrence
12 years after surgery. Swelling was the most frequent clinical manifestation (43 patients, 64%). Almost all were hirsute (65 patients,
97%). Clinical presentation included pilonidal abscess treated by
drainage (22 cases, 33%); chronic discharge (32, 48%); and single
uninfected sinus (13, 19%).
Surgical technique
Introduction
There are many ways to treat pilonidal sinus, perhaps reflecting the overall unsatisfactory results. Pilonidal sinus
or jeep disease [1] is common and afflicts young patients
after puberty with a male-female ratio of 3 : 1, it is rare afG. Milito () 1 F. Cortese C. U. Casciani
Cattedra di Clinica Chirurgica,
Universit di Roma Tor Vergata, Ospedale SantEugenio,
10 Piazzale dellUmanesimo, I-00144 Roma, Italy
Present address:
1
1530/b Via Cassia, I-00123 Roma, Italy
114
AB = BC = CD = DA = AE = EF = AC = AF
ABC = BAC = BCA = AEF = 60
AI = LC = AH = GF
EM = BN
E
H
M
G
A2
C
A1
AC
BD
8
7.5
7
6.5
6
5.5
5
4.5
4
3.5
3
13.80
12.94
12.07
11.21
10.35
9.49
8.62
7.76
6.9
6.04
5.17
FGMHA
FE
A INLC
2
AE
References
Fig. 1 Marking of a rhombus, including the pilonidal sinus
discharged with instructions about personal hygiene and the necessity for repeated shaving of the surrounding skin. All patients were
seen after 2 weeks and then after 3, 6, 8, and 12 months after discharge.
Results
Discussion
115
16. Lord PH, Millar DM (1965) Pilonidal sinus: a simple treatment.
Br J Surg 52: 298300
17. Bascom JU (1987) Repeat pilonidal operations. Am J Surg
154: 118122
18. Dufourmentel C (1963) An L shaped flap for lozenge shaped
defects. Transactions of the Third International Congress of
Plastic Surgery. Excerpta Medica Foundation, Amsterdam,
pp 722726
19. Azab ASG, Kamal MS, Saad RA, Abou al Atta KA, Ali NA
(1984) Radical cure of pilonidal sinus by a transposition rhomboid flap. Br J Surg 71: 154155
Springer-Verlag 1998
O R I G I N A L A RT I C L E
Abstract Hysterectomy is associated with severe constipation in a subgroup of patients, and an adverse effect on
colonic motility has been described in the literature. The
onset of irritable bowel syndrome and urinary bladder dysfunction has also been reported after hysterectomy. In this
prospective study, we investigated the effect of simple
hysterectomy on ano-rectal physiology and bowel function.
Thirty consecutive patients were assessed before and 16
weeks after operation. An abdominal hysterectomy was
performed in 16 patients, and a vaginal procedure was performed in 14. The parameters measured included the mean
resting, and maximal forced voluntary contraction anal
pressures, the recto-anal inhibitory reflex, and rectal sensation to distension. In 8 patients, the terminal motor
latency of the pudendal nerve was assessed bilaterally.
Pre-operatively, 8 patients were constipated. This improved
following hysterectomy in 4, worsened in 2, and was unchanged in 2. Symptomatology did not correlate with
changes in manometry. Although, the mean resting pressure was reduced after hysterectomy (57 mmHg
53 mmHg, P = 0.0541), the maximal forced voluntary contraction pressure was significantly decreased (115 mmHg
105 mmHg, P = 0.029). This effect was more pronounced
in those with five or more previous vaginal deliveries
(P = 0.0244, n = 9). There was no significant change in the
number of patients with an intact ano-rectal inhibitory reflex after hysterectomy. There was no change in rectal sensation to distension, and the right and left pudendal nerve
terminal motor latencies were unaltered at follow-up. Our
results demonstrate that hysterectomy causes a decrease in
the maximal forced voluntary contraction and pressure, and
this appears to be due to a large decrease in a small group
of patients with previous multiple vaginal deliveries.
Introduction
Hysterectomy is the second most frequently performed major surgical procedure in the developed world [1], and although most operations are uneventful, the complication
rate during hospital admission is approximately 7% [2, 3].
Aside from the initial complications, post-hysterectomy irritable blowel syndrome develops de novo in 13% of pa-
117
tients at 6 weeks [4]. A small group of patients also proceed to develop idiopathic slow-transit constipation [5].
This is believed to be the result of damage to the autonomic
nerve supply of the hindgut, resulting in functional obstriction [6], and may necessitate left hemicolectomy [7]. Furthermore, altered relaxation of the internal anal sphincter
and decreased rectal sensation have been described following radical hysterectomy for carcinoma of the cervix [8].
Therefore, in order to evaluate fully the effect on ano-rectal physiology, we examined a group of patients, before
and after simple abdominal and vaginal hysterectomy.
Results
The median age was 46 years (range 30 64). Sixteen patients had an abdominal, and fourteen had a vaginal hysterectomy, with preservation of ovarian function. The most
frequent indications for operation were abnormal bleeding
(40%), chronic pelvic pain (30%) and leiomyomas (14%).
Age showed a negative correlation with mean resting anal
pressure (correlation coefficient, r = 0.4241; P = 0.019),
but not with maximal forced voluntary contraction anal
pressure (correlation coefficient, r = 0.24; P = 0.19). The
patiens who had a vaginal hysterectomy were significantly
older (mean age 44 years) than those who had an abdominal hysterectomy (mean age 54 years). However, there was
no difference between the groups regarding the number of
vaginal deliveries, or the mean resting, or forced voluntary
contraction anal pressures.
Seventeen patients reported an unchanged bowel pattern at follow-up. In nine, there was a slight increase in the
frequency of bowel motions, and a decrease in four. Only
one patient required stool softening agents post-operatively.
The mean resting anal pressure was unchanged by hysterectomy, but the forced voluntary contraction anal pressure was significantly reduced at 16 week (115 mmHg
105 mmHg, P = 0.029). This effect was due to a large decrease in nine patients with a history of five or more vaginal deliveries. In this group, the mean drop in forced
voluntary contraction pressure after hysterectomy was
21 mmHg (112 mmHg 91 mmHg, P = 0.048). In those
who had had fewer than five vaginal deliveries, the mean
drop in forced voluntary contraction pressure was 3 mmHg
(115 mmHg 112 mmHg, not significant). There was no
difference between these two groups regarding the age of
the patients or the number who had a vaginal hysterectomy
(4/9 and 10/21), or those who had an abdominal hysterectomy (5/9 and 11/21). The number of vaginal deliveries
did not correlate with the mean resting (correlation coefficient, r = 0.21) or maximal forced voluntary contraction
pressures (correlation coefficient, r = 0.027).
The threshold volume and the urge volume were unchanged at follow-up. The terminal motor latency of the
pudendal nerve was not effected by hysterectomy, and no
difference was found in the number of patients with an intact recto-anal inhibitory reflex.
Statistical analysis
Correlation coefficients were calculated by linear regression analysis and ANOVA. Paired, and unpaired, Students
t-tests were used to compare variables between the various groups, and significance was set at P 0.05.
Discussion
Pelvic surgery can damage the autonomic innervation of organs leading to dysfunction of the urinary bladder [9] and,
in a small group of patients, to intractable constipation [5,
6]. Hysterectomy has been reported to result in increased
rectal sensitivity [4], and impaired motility of the distal sigmoid colon has also been described [7]. In association with
abnormalities of bladder function, this is most likely due to
damaged fibres of the inferior hypogastric plexus.
It is believed that traction injury to the parasympathetic
nerves (S2, S3 and S4) lying lateral to the vaginal fornix,
and in the broad ligament of the uterus, is responsible for
post-hysterectomy constipation [10]. Although cases of faecal incontinence and rectal prolapse have been reported [11],
118
no adverse effects on the pudendal nerve-innervated, external and sphincter mechanism have yet been described [12].
In this study, we found a significant decrease in the maximal forced voluntary contraction pressure, generated by
the external and sphincter, at 16 weeks. This was due to
the large decrease recorded for nine patients who had had
five or more previous vaginal deliveries. The mechanism
responsible may be direct trauma to the external and
sphincter at the time of operation, or damage to the S3 and
S4 nerves supplying the puborectalis part of the external
sphincter. It is worth noting that the external urethral
sphincter, which is innervated by the pudendal nerve, is
particularly susceptible to injury following hysterectomy
[9, 13]. In this study, no patient reported urinary incontinence, and we found no difference in the terminal motor
latency of the pudendal nerve at follow-up. The resting anal
pressure in our study showed a negative correlation with
age, and in contrast to previous reports, was unchanged
following hysterectomy [8].
In the past, hysterectomy has been associated with the
onset of irritable bowel syndrome in up to 13% of cases
[4], and we found a significant change in bowel pattern at
16 weeks in 13 of our patients. In agreement with previous reports, rectal sensitivity was unchanged following
hysterectomy [10]. It has been suggested that hysterectomy
may have a greater effect on the act of defecation than on
colonic motility, as up to 25% of patients report increased
straining at stool at follow-up [14]. No patient in this study
developed faecal incontinence and the number of patients
with an intact recto-anal inhibitory reflex remained the
same at follow-up.
Although no patient in this study developed faecal incontinence, we believe that hysterectomy may have an adverse effect on the external sphincter mechanism in a subgroup of patients. It appears that patients with a history of
multiple vaginal deliveries are more prone to external
sphincter injury after hysterectomy. The mechanism remains unclear, but the terminal motor latency of the pudendal nerve seems to be unaffected. This appears to indicate either direct trauma to the sphincter itself, or to the
nerves supplying the puborectalis.
In summary, we have demonstrated that following hysterectomy there is a decrease in the maximal pressure gen-
erated by the external anal sphincter. This effect is pronounced in a small group of patients with previous multiple vaginal deliveries. Although this decrease did not have
any adverse effect at 16 weeks, it may have a detrimental
effect on those patients with an already compromised
sphincter.
References
1. Graves EJ (1992) National hospital discharge survey: annual
summary, 1990. Vital and health statistics, series 13, no. 112.
National Centre for Health Statistics, Hyattsville, Maryland
2. Carlson KJ, Miller BA, Fowler FJ (1994) The Maine womens
health study outcomes of hysterectomy. Obstet Gynecol
83: 556 564
3. Clarke A, Black N, Rowe P, Mott S, Howle K (1995) Indications
for and outcome of total abdominal hysterectomy for benign
disease: a prospective cohort study. Br J Obstet Gynaecol
102: 611 620
4. Prior A, Stanley KM, Smith ARB, Read NW (1992) Relation
between hysterectomy and the irritable bowel: a prospective
study. Gut 33: 814 817
5. Roe AM, Bartolo DCC, McMortensen NJ (1988) Slow transit
constipation comparison between patients with or without previous hysterectomy. Dig Dis Sci 33: 1159 1163
6. Varma JS (1992) Autonomic influences on colorectal motility
and pelvic surgery. World J Surg 16: 811 819
7. Vierhout ME, Schreuder HWB, Veen HF (1993) Severe slow
transit constipation following radical hysterectomy case report. Gynecol Oncol 51: 401 403
8. Barnes W, Waggoner S, Delgado G, et al (1991) Manometric
characterisation of rectal dysfunction following radical hysterectomy. Gynecol Oncol 42: 116 119
9. Taylor T, Smith AN, Fulton PM (1990) Effects of hysterectomy
on bowel and bladder function. Int J Colorectal Dis 5: 228
10. Devrode G, Lamarche J (1984) Functional importance of extrinsic parasympathetic innervation of the distal rectum and colon
in man. Gastroenterology 86: 287
11. Schwartz S (1994) Textbook of surgery. 6th edn. McGraw-Hill,
New York, p 1220
12. Prior A, Stanley K, Smith ARB, et al (1992) Effect of hysterectomy on anorectal and urethrovesical physiology. Gut 33: 264
267
13. Bartolo DCC, Jarratt JA, Read MG, Donnelly TC, Read NW
(1983) The role of partial denervation of the puborectalis in idiopathic faecal incontinence. Br J Surg 70: 664 667
14. Heaton KW, Parker D, Cripps H (1993) Bowel function and irritable bowel symptoms after hysterectomy and cholecystectomy a population based study. Gut 34: 1108 1111
Springer-Verlag 1998
O R I G I N A L A RT I C L E
P. J. Hainsworth D. C. C. Bartolo
Introduction
Omission of a temporary loop ileostomy in selected patients undergoing restorative proctocolectomy is not new.
First advocated more than 10 years ago [1], avoidance of
an ileostomy remains controversial. Perceptions of an
easier, quicker and more reliable anastomosis with stapled
versus hand-sewn construction may account for the increased popularity of stoma avoidance [2]. Opponents cite
the relative safety of a temporary ileostomy [3] and express concerns about a possible increase in frequency of
120
Table 1 Published series of
restorative proctocolectomy
with ileostomy (I) and with no
ileostomy (NI). (Numbers in
parentheses represent I group).
All complications shown as
percentages. (Sel selected,
Mat matched controls,
Rand randomised, Con consecutive, Hist historic controls)
Ref/Year
1. 8083
4. 8190
5. 8488
6. 8388
7. 8891
8. 8489
9. 8788
10. 8789
11. 8991
12. 8991
13. 8993
14. 8993
15. 8292
Present
a
b
c
d
e
Numbera
NI (I)
21
37 (37)
38
32 (53)
22 (23)
29 (35)
16 (15)
25 (21)
30 (28)
50 (50)
68 (63)
68
71 (87)
72 (30)
Study
designb
Sel
Sel/Mat
Sel
Sel
Rand
Sel
Con (Hist)
Con (Hist)
Sel
Sel (Mat)
Sel (Hist)
Sel
Sel
Sel
IPAAc
Hand
Hand
Stapled
Both
Stapled
Hand
Hand
Hand
Stapled
Stapled
Stapled
Stapled
Stapled
Both
Bowel
obstruction
Pouch
faile
10
8 (14)
11
18 (53)
5 (9)
17 (11)
6 (0)
4 (0)
10 (11)
18 (6)
15 (24)
5
30 (13)
6 (3)
19
?
?
6 (23)
9 (22)
7 (14)
13 (13)
24 (16)
7 (29)
0 (2)
7 (10)
?
?
8 (3)
10
3 (8)
0
6 (17)
5 (0)
?
0 (0)
4 (0)
0 (0)
0 (0)
0 (19)
3
1 (3)
0 (0)
pelvic sepsis or more serious consequences if sepsis occurs. Table 1 shows the salient findings from reports comparing patients with and without temporary diversion.
In 1990 we reported a series of ileostomy closures after restorative proctocolectomy and found the morbidity to
be low with most patients able to leave hospital soon after
the procedure [16]. Therefore, we initially adopted a very
liberal approach to the use of temporary ileostomies. Between 1990 and 1995 a stoma was increasingly avoided
providing the operation proceeded satisfactorily and that
experience forms the basis of this report.
without a proximal stoma. A temporary ileostomy was avoided providing the patient was not acutely unwell (fever, tachycardia, abdominal tenderness, toxic dilatation) and not malnourished. Further criteria for stoma avoidance included performance of a straightforward
operation with creation of a sound, tension-free anastomosis. Confidence in omitting an ileostomy increased during the study period.
Recent steroid intake or immunosuppression did not dictate the need
for a stoma.
Median follow-up was 17 months (051 months). Outpatient follow-up at regular intervals assessed 24-hour stool frequency, nocturnal emptying, continence and use of constipating medication.
Results
Patients and methods
Between June 1990 and March 1995, 102 patients (57 male, 45 female) underwent restorative proctocolectomy. Age ranged between
12 and 76 (median 36) years. Preoperative diagnoses were ulcerative colitis (n = 83); indeterminate colitis (n = 4); familial adenomatous polyposis (FAP) (n = 5); idiopathic megacolon and/or slow transit constipation (n = 6); Hirschsprungs disease (n = 1); diffuse angiodysplasia (n = 1) and colon cancer (n = 2).
Patients who were severely ill underwent colectomy and ileostomy with a view to delayed reconstructive surgery. Other patients
underwent immediate restorative proctocolectomy or, if the colectomy had been carried out elsewhere, rectal excision and reconstruction.
All operations were performed or directly supervised by the senior author (D. C. C. B.). Broad-spectrum antibiotics and subcutaneous heparin were routinely given. The small bowel was mobilised to
the origin of the superior mesenteric artery, preserving the ileal
branch of the ileocolic artery. The preferred method of pouch construction was the stapled J with 20 cm limbs. Early on in the series, W pouches were created in 7 patients. The pouch-anal anastomosis was single-stapled in 91 patients preserving 12 cm of mucosa above the dentate line. The remaining 11 patients underwent
mucosectomy with hand-sewn anastomosis to the dentate line.
Those in Group I (temporary ileostomy) were defunctioned at a
convenient point proximal to the pouch. Group NI (no ileostomy)
patients underwent restorative proctocolectomy or proctectomy
A temporary loop ileostomy was added at the time of fashioning the pouch in 30 out of 102 patients (29%) (Table 2).
All those with toxic megacolon or acute colonic bleeding
underwent initial subtotal colectomy with delayed pouch
construction. Among 25 other patients with acute colitis,
7 underwent immediate restorative proctocolectomy, three
without a covering ileostomy. All those undergoing restorative proctocolectomy for chronic relapsing colitis, dysplasia or adenomatous polyps had immediate pouch construction, mostly without a covering stoma. Patients undergoing pouch surgery without an ileostomy were contrasted
with those given an elective ileostomy (Table 3). There
were no significant differences between the groups although trends towards low body weight and low serum albumin were present in Group I.
A late stoma was required by 6 out of 72 patients (8%)
in Group NI for the following reasons: pouch anastomotic
leak (2 cases), pelvic sepsis, small bowel perforation,
pouch-vaginal fistula and one instance of staple gun
sphincter injury. None of these six cases had acute colitis
at the time of creating the pouch. One elderly male patient
who suffered a leak from the top of the pouch was taking
121
Table 2 Use of diverting ileostomy at time of pouch procedure: no
ileostomy (NI); proximal ileostomy (I)
Indication for pouch procedure
NI (n=72)
I (n=30)
34
6
3
10
19
0
10
1
4
5
9
1
Age (years)
Sex
Weight (kg)
History (years)
Hb
WBC
Serum albumin
On steroids
median (range)
M:F
median (range)
median (range)
<110 g/l
12 109/l
<36 g/l
NI (n=72)
I (n=30)
39 (1265)
41 : 31
68 (44105)
5 (025)
9 (13%)
7 (10%)
6 (8%)
22 (31%)
32.5 (1576)
16 : 14
60.5 (4895)a
4 (024)
4 (13%)
4 (13%)
7 (23%)b
13 (43%)
I (n=30)
2a
2 (1a)
1a
1
0
0
1a
1
1
2
1
1
1a
0
1
0
3
3
1
1
13
1
0
1
0
8
0
0
0
1
1
1
a
a
Delayed ileostomy
All late events
After pouch procedure or closure of any ileostomy
d
Including late ileostomies
b
c
Table 5 Pouch function at intervals after establishing intestinal continuity: no ileostomy (NI); proximal ileostomy (I)
Time after
surgery
NI (n=72)
I (n=30)
Freq/24 h
Med (range)
6 weeks
6 months
12 months
7 (220)
5 (120)
4 (211)
Noctural
emptying
6 weeks
6 months
12 months
42/67 (63%)
16/55 (29%)
9/42 (21%)
16/27 (59%)
6/23 (26%)
7/21 (33%)
Soiling
6 weeks
6 months
12 months
30/68 (44%)
19/54 (35%)
11/43 (26%)
12/28 (43%)
5/23 (22%)
6/21 (29%)
7 (312)
5 (310)
5 (210)
122
Discussion
Numerous reports indicate that restorative proctocolectomy may be safely undertaken without a covering ileostomy in selected patients [48, 10, 11, 13, 14]. Potential
benefits of stoma avoidance include the need for just one
operation, reduced operating time, shorter hospital stay and
avoidance of ileostomy related complications [4, 8, 10, 11,
13, 17]. The crux of the argument for and against temporary ileostomy rests on the relative incidence of complications and their severity in each group.
All available studies are limited by a variable degree of
patient selection bias. The only randomised study excluded
patients taking steroids [7] and in the 45 low-risk patients
who were randomised, only two cases of pelvic sepsis occurred. Even in one apparently consecutive series, the authors excluded acute and emergency cases [10]. Others
have included the vast majority of patients although those
with acute fulminant colitis are usually excluded [13]. One
study apparently included patients with acute fulminant
colitis [14].
In selecting patients for stoma avoidance, Galundiuk et
al. [4] stress the importance of an absolute lack of tension
on the anastomosis, good blood supply to the terminal ileum, good general health and absence of recent steroid intake. In the present series, fewer patients were on steroids
in Group NI (31% vs 43%) but most serious complications
occurred in those not taking steroids. The literature is divided on the risks of steroids in those without a covering
stoma and although several series show no increase in complications [1012, 17], one study reports an increase in pelvic sepsis with 20 mg/day prednisolone [12].
Because of the potentially disastrous consequences of
pelvic sepsis and the relative safety of creating and closing a temporary ileostomy, many consider temporary diversion to be preferable [3, 12, 1820]. The overall incidence of pelvic sepsis among 1218 patients treated at the
Majo clinic was 5% [21]. The collected data in Table 1
show no obvious increase in pelvic sepsis or pouch failure
in those without ileostomies. However, if an anastomotic
leak occurs, are the consequences worse in the absence of
a proximal stoma? In this series, pelvic sepsis occurred in
4/72 patients in Group NI, three of whom required late ileostomies. In Group I, one of 30 patients developed a leak
complicated by a pelvic abscess, fistula and anastomotic
123
References
1. Thow GB (1985) Single-stage colectomy and mucosal proctectomy with stapled antiperistaltic ileoanal reservoir. In: Dozois RR
(ed) Alternatives to conventional ileostomy. Year Book Medical
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2. Winslet MC, Barsoum G, Pringle W, Fox K, Keighley MRB
(1991) Loop ileostomy after ileal pouch-anal anastomosis is it
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(1994) Loop ileostomy for temporary fecal diversion. Am J Surg
167: 519522
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34: 870873
5. Peck DA (1988) Stapled ileal reservoir to anal anastomosis. Surg
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6. Hosie KB, Grobler SP, Keighley RMB (1992) Temporary loop
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7. Grobler SP, Hosie KB, Keighley MRB (1992) Randomized trial
of loop ileostomy in restorative proctocolectomy. Br J Surg
79: 903906
8. Everett WG, Pollard SG (1990) Restorative proctocolectomy
without temporary ileostomy. Br J Surg 77: 621622
9. Jrvinen HJ, Luukkonen P (1991) Comparison of restorative
proctocolectomy with and without covering ileostomy in ulcerative colitis. Br J Surg 78: 199201
10. Matikainen M, Santavirta J, Hiltunen K (1990) Ileoanal anastomosis without covering ileostomy. Dis Colon Rectum 33:
384388
11. Sagar PM, Lewis W, Holdsworth PJ, Johnston D (1992) Onestage restorative proctocolectomy without temporary defunctioning ileostomy. Dis Colon Rectum 35: 582588
12. Tjandra JJ, Fazio VW, Milsom JW, Lavery IC, Oakley JR, Fabre
JM (1993) Omission of temporary diversion in restorative proctocolectomy is it safe? Dis Colon Rectum 36: 10071014
13. Sugerman HJ, Newsome HH (1994) Stapled ileoanal anastomosis without a temporary ileostomy. Am J Surg 167: 5866
14. Mowschenson PM, Critchlow JF (1995) Outcome of early surgical complications following ileoanal pouch operation without
diverting ileostomy. Am J Surg 169: 143145
15. Cohen Z, McLeod RS, Stephen W, Stern HS, OConnor B, Reznick R (1992) Continuing evolution of the pelvic pouch procedure. Ann Surg 216: 506511
16. Lewis P, Bartolo DCC (1990) Closure of loop ileostomy after
restorative proctocolectomy. Ann R Coll Surg Engl 72: 263265
17. Jrvinen HJ, Luukkonen P (1993) Coloproctectomie et anastomose ilo-anale en un temps: bnfices et facteurs de risques.
Ann Chir 47: 971975
18. Wong WD, Rothenberger DA, Goldberg SM (1985) Ileoanal
pouch procedures. Curr Probl Surg 22 (3): 978
19. Wexner SD, Taranow DA, Johansen OB, Itzkowitz F, Daniel N,
Nogueras JJ, Jagelman DG (1993) Loop ileostomy is a safe option for fecal diversion. Dis Colon Rectum 36: 349354
20. Metcalf AM, Dozois RR, Kelly KA,Wolff BG (1986) Ileal
pouch-anal anastomosis without temporary ileostomy. Dis Colon Rectum 29: 3335
21. Pemperton JH (1985) Complications, management, failure and
revisions. In: Nicholls RJ, Bartolo D, Mortensen N (eds) Restorative proctocolectomy. Blackwell Scientific Oxford pp 3452
22. Groom JS, Nicholls RJ, Hawley PR, Phillips RKS (1993) Pouchvaginal fistula. Br J Surg 80: 936940
Springer-Verlag 1998
O R I G I N A L A RT I C L E
Abstract Dyschezia may be caused by pelvic floor dyssynergia, which takes place when a paradoxical contraction or a failure to relax the pelvic floor muscles occurs
during attempts to defecate. The aim of our study was to
set up a new bimodal rehabilitation programme for pelvic
floor dyssynergia, which combined pelviperineal kinesitherapy and biofeedback, and to evaluate the results of this
treatment. Thirty-five patients (age range: 2864 years;
mean age: 42.5 years) from the outpatient unit of the Clinica Chirurgica of the University of Florence, Italy, and an
age-matched group of 10 healthy control subjects (age
range: 3159 years; mean age 45.7 years) with normal
bowel habits and without any defecatory disorders, were
studied. The 35 patients were symptomatic for dyschezia
without slow colonic transit and had been diagnosed as being affected by pelvic floor dyssynergia. No evidence of
any organic aetiology was present but all demonstrated
both manometric and radiological evidence of inappropriate function of the pelvic floor. All of the patients underwent bimodal rehabilitation, using the combined training
programme Clinical evaluation, computerized anorectal
manometry and defecography were carried out 1 week before and 1 week after a completed course in bimodal rehabilitation. The control group underwent manometric and
defecographic examination. Their results were compared
with those of the 35 patients before and after training. After the programme, all 35 patients had a very significant
increase in stool frequency (P<0.001), while laxative and
enema-induced bowel movements had become significantly less frequent (P<0.001). After bimodal rehabilitation, computerized anorectal manometry showed some peF. Pucciani () A. Bologna F. Cianchi C. Cortesini
Istituto di Clinica Chirurgica Generale e Discipline Chirurgiche,
Universit degli Studi di Firenze, Unit di Coloproctologia (UCP),
Viale Morgagni 85, I-50134 Firenze, Italy
M. L. Rottoli
Radiodiagnostica 1, Dipartimento Fisiopatologia Clinica,
Universit degli Studi di Firenze, Italy
S. Forconi M. Cutell
U.O. Riabilitazione, Ospedale Mauriziano, Torino, Italy
125
Introduction
Pelvic floor dyssnergia may be defined as a faecal evacuation disorder which is a consequence of some functional
outlet obstruction [1]. It is characterized by a paradoxical
contraction or failure to relax the pelvic floor muscles during attempts at defecating. Clinical manifestations can include straining, feeling of incomplete evacuation and/or
the need to digitally evacuate the rectum [2]. D. M. Preston and J. E. Lennard-Jones [3] suggested that some form
or retraining might help patients to relax the striated muscles of the pelvic floor during defecation. Biofeedback appears to be effective for this form of dyschezia [4], even
though the questions of how, when and why it works remain unanswered [5]. Pelviperineal kinesitherapy, which
has been tried occasionally in cases of dyschezia [6], is a
specific muscular re-education technique for the pelvic
floor muscles [7].
The aims of this study were to set up a new bimodal rehabilitation technique using both pelviperineal kinesitherapy and biofeedback, for pelvic floor dyssynergia,
and to evaluate the results of this treatment.
126
Computerized anorectal manometry
Anorectal manometry was performed using standard techniques [8].
Recordings and analyses of the tracings were made using a computerized system (Dyno System, Menfis s.r.l., Bologna, Italy) as previously described [9].
Anal Resting Pressure (ARP) was recorded in mmHg with the
stationary pull-through technique and the computer identified the
maximal pressure (Pmax), the mean pressure (Pm) and the high pressure zone area (HPZ area), where HPZ area was = (Pi Li): Pi was
the pressure value in mmHg at each sampling, and Li was the length
in millimeters between two successive samplings. The maximal voluntary contraction (MVC) was examined by evaluating the voluntary contractions of the anal sphincter; amplitude was expressed in
mmHg, duration in seconds. The rectoanal inhibitory reflex (RAIR)
was elicited by inflating a soft balloon in the rectum at 10 cm from
the anal verge wiith 40 ml of air, our normal value to induce complete relaxation [9]. The computer quantified the total duration of reflex (TDR) in seconds: TDR was equal to the complete amount of
relaxation time (RT) in seconds plus contraction time (CT) in seconds, as suggested by Martelli [10]. The computer also quantified
the maximal amplitude of relaxation (MAR) expressed in percent,
the residual pressure at the lowest point of the RAIR (Pres in mmHg),
the mean RAIR pressure (Pm RAIR in mmHg) and the area of the reflex where the RAIR area was = (Pi Li): Pi was the pressure value in mmHg at each sampling and Ti was the time in seconds between
two successive samplings.
The first distension volume at which internal anal sphincter relaxation had occurred, i.e. the RAIR threshold, (RAIRT), and the distension volume at which an initial transient sensation had taken place,
i.e. the conscious rectal sensitivity threshold, (CRST), were determined in all patients and controls. The maximal tolerated volume
(MTV) was also measured in all subjects and it was considered an
expression of rectal reservoir capacity. Compliance of the rectum
was expressed by the ratio mmHg/ml of inflated air, measured by
means of the pressure/volume curve. Manometric signs of PFD were
high anal canal pressure (Pm; HPZ area) and impaired RAIR, i.e. incomplete relaxation with a short duration of the reflex [9].
Defecography
All patients and controls underwent defecography according to the
methods suggested by the Italian Working Team [11]. The radiological assessment was carried out at rest, during contraction, and during expulsion of the barium. The anorectal angle (ARA) was measured between the longitudinal axis of the anal canal and the tangential line to the posterior rectal wall, and was expressed in degrees.
The pelvic floor descent (PFDe), which was defined as the vertical
distance between the pubococcygeal line and the anorectal junction,
was expressed in millimetres. A qualitative evaluation, diagnostic
for pelvic floor dyssynergia, was made by noting the persistence of
the puborectalis indentation during evacuation.
Bimodal rehabilitation
All patients underwent bimodal rehabilitation. A single cycle consisted of ten outpatient sessions. Each session lasted 1 hour and took
place twice a week. The first step was pelviperineal kinesitherapy.
This was then combined with biofeedback training from the fifth session until the end of the cycle.
From the start to the end of treatment, patients were asked to
record stool frequency. They were also asked to indicate whether laxatives and/or enema assistance had been required.
a) Pelviperineal kinesitherapy
The cycle of pelviperineal kinesitherapy followed the standard sequence listed below, but was adapted to the individual woman. During each session two essential steps were taken: the exercises of the
preliminary lesson on relaxed breathing and corporeal consciousness (used at the start of all sessions),
diaphragmatic breathing,
marking of perineal area, made easier by peri- and
intra-anal digital manipulation,
location and focusing of agonist, antagonist and synergic muscles on the perineal plane.
2nd session:
127
Table 1 Clinical evaluation
Controls
(10)
Patients
Patients
(35) (pre) (35) (post)
6.1 1.2
2.7 1.8 a
1.2 0.3 a
1.6 0.6 a
26/35
8/35
6.3 2.0
0.4 0.2
0.6 0.1
3/35
Results
Clinical symptoms
Defecography
Table 1 shows a comparison of clinical characteristics of
controls and patients.
All 35 patients had a very significant increase in bowel
movement frequency (P < 0.001) after bimodal rehabilitation. The frequency of laxative-induced (P < 0.001) and enema-induced (P < 0.001) bowel movements was significantly reduced, and the need to digitally evacuate the rectum (74.2% of our patients had used this technique) was
no longer necessary after the therapeutic treatment. Only
3 women still had obstructed micturition at the end of the
ten sessions.
Computerized anorectal manometry
After bimodal rehabilitation, anal canal pressure had increased, but not significantly. Maximal pressure (P < 0.01),
mean pressure (P < 0.01), and HPZ area (P < 0.05) were significantly higher than those of the controls (Table 2). After treatment, MVC had no significant differences in amplitude when compared with pre-treatment values. MVC
Table 2 Anal canal pressures
Pmax
mmHg
Controls
Patients (pre)
Patients (post)
a
b
c
78.0 10.2
97.1 11.1 b
110.2 17.4 b
Discussion
HPZ area
(Pi Li)
1678.4 279.3
2685.1 374.3 a
2871.6 178.9 a
MVC
P (mmHg)
T (s)
124.1 3.7
151.1 15.6 a
163.7 20.4 a
24.3 7.3
17.3 5.1 c
24.5 6.8
128
Table 3 RAIR
Controls
Patients (pre)
Patients (post)
a
b
c
RT (s)
CT (s)
TDR (s)
Pm
mmHg
Pres
mmHg
MAR
(%)
RAIR area
(P Ti)
9.1 0.2
7.8 1.6
7.7 3.1
20.4 2.5
12.5 0.3 c
19.9 4,8
29.5 2.7
20.2 1.9 b
26.6 8.7
22.6 2.4
15.9 9.8 a
19.4 7.3
1.3 0.7
12.0 3.7 c
2.1 0.4
97.4 2.2
79.0 3.0 c
96.8 3.1
654.7 39
365.1 32 c
612.6 56
Controls
Patients (pre)
Patients (post)
a
b
ARA (degrees)
94 3
85 6 a
87 2 a
110 3
87 4 b
106 9
39.4 11
32.6 18
37.6 15
69.4 11
49.5 12 a
82.3 13 a
Barium
trapping (%)
Puborectalis indentation
(n patients/total patients)
10
50
25
35/35
mas had decreased (P < 0.001). The need to digitally evacuate the rectum disappeared and only three patients showed
obstructed micturition, with hesitancy. These symptomatic
improvements were confirmed by manometric-proctographic data.
After treatment, manometric results showed that anal
canal pressure was higher (Table 2) and that there was a
normal rectoanal inhibitory reflex (Table 3). Anal resting
pressure was statistically higher than that of the controls,
but it was not significantly different from pre-treatment
values. This was also shown by the defecographic resting
ARA which remained unchanged and was statistically
more narrow than that of controls (P < 0.05). It is difficult
to say what might have been the functional mechanism
which determined the anal canal hypertonia. The failure of
anorectal myectomy to resolve anismus [3], the absence of
any anal fissure, and recent advances in morphological
evaluation using sonography [13] suggest that the internal
anal sphincter cannot be responsible. On the other hand,
since the influence of striated sphincter muscles on resting
anal tone values is well known (1530%) [14, 15], it is possible that stronger contractions of the puborectalis muscle
and the external anal sphincter might have induced the increased anal canal pressure. But we do not know what the
aetiological factor is. Furthermore, the fact that there were
minimally higher changes in ARP (Table 2) with increased
anal squeeze pressure has also been reported by others after the use of biofeedback [5]. Nevertheless, anal hypertonia does not seem to be an important factor for defecation
retraining if correct pelvic floor function can be obtained.
Anal canal hypertonia is a physical and/or manometric sign
which indicates increased anal sphincteric strength: it does
not offer any information on anal sphincteric muscle coordination. Such strength is only one aspect of anal function.
On the contrary, the coordination of agonist, antagonist and
synergic muscles of the pelviperineal plane is the crucial
129
Fig. 1 P1010 = manometric
channel. t1; t2 = manometric
sample window: times at the
start (t1), at the end (t2). RAIR
elicited: r/40 = inflation of balloon 40 ml air. Upper tracing:
normal subject at r/40 (empty
arrow). Lower tracing: patient
with pelvic floor dyssynergia.
Note the high excitatory response at r/40 (full arrow) and
the incomplete relaxation during reflex
130
References
1. Badiali D, Habib FI, Corazziari E, Viscardi A, Primerano L, Anzini F, Torsoli A (1991) Manometric and defaecographic patterns of straining. J Gastroint Mot 3: 171
Springer-Verlag 1998
O R I G I N A L A RT I C L E
Abstract Purpose: The aetiology of idiopathic megarectum and idiopathic megacolon is unknown. A previous study in patients with chronic idiopathic intestinal
pseudo-obstruction, a condition also associated with a dilated gut, identified the possible involvement of herpes
viruses. This study therefore aimed to determine whether
these viruses may also be implicated in the pathogenesis
of these conditions. Methods: Resected large bowel from
three patients with idiopathic megarectum and three patients with idiopathic megacolon were studied. Histology
for viral inclusions and nested polymerase chain reaction
(PCR) using specific primers for cytomegalovirus, Epstein-Barr virus, herpes simplex virus type 1 and varicella
zoster virus was performed. DNA was extracted from paraffin-embedded blocks by proteinase K and phenol chloroform extraction. Results: Viral inclusions were not
seen. PCR failed to identify DNA of the four herpes viruses tested. Conclusion: Patients with idiopathic megarectum or idiopathic megacolon may have subtle abnormalities of the enteric innervation, but these do not appear to be attributable to the neurotropic effects of the
herpes viruses studied.
Key words Idiopathic megarectum Megacolon
Herpes DNA viruses
Rsum But: Ltiologie du mgarectum idiopathique et
du mgaclon idiopathique est inconnue. Une tude pralable chez des patients avec une pseudo-obstruction intestinale idiopathique chronique, une condition associe avec
une dilatation du clon, a montr le rle potentiel dune atteinte virale herptique. Cette tude a t entreprise pour
dterminer si ces virus peuvent galement tre impliqus
J. M. Gattuso H. S. Debinski M. A. Kamm ()
St. Marks Hospital, Northwick Park,
Watford Road, Harrow,
Middlesex HA1 3UJ UK
H. O. Kangro D. Jeffries
Department of Virology,
St. Bartholomews Hospital,
London, UK
132
crofuge at 13 000 rpm for 10 min and the xylene discarded. The resulting pellet was rinsed in 95% ethanol and spun at 13 000 rpm for
a further 10 min. This was repeated with clean ethanol to ensure complete removal of the xylene. The pellet was dried in a heating block
at 45 C, resuspended in sterile distilled water and incubated with 0.5
mg/ml proteinase K at 37 C for 12 h. Phenol chloroform was added to the samples, which were mixed by inversion and centrifuged
at 13 000 rpm for 15 min. The phenol layer was discarded and 0.3 M
sodium acetate, 3 l of tRNA and ethanol was added to the aqueous
phase. This was mixed and left to precipitate at 70 C for 20 min.
The samples were spun for 15 min at 4 C at 13 000 rpm to pellet the
DNA. The pellet was washed in 70% ethanol, centrifuged for 5 min
at 13 000 rpm, dried, resuspended in sterile distilled water and boiled
for 15 min.
The PCR was performed as described by Saiki [9] using recombinant DNA polymerase from Thermus aquaticus (Amplitaq; Perkin-Elmer Cetus) according to the manufacturers protocol. All preparative work for the PCR was carried out in an ultraviolet-irradiated class II microbiological safety cabinet, using autoclaved plastic
tubes and pipette tips throughout.
Amplification reactions were carried out in a total volume of
100 l comprising 50 l test sample and 50 l reaction buffer in
0.6-ml microcentrifuge tubes. The reaction buffer was prepared in
bulk mastermix at double strength to give final concentrations of
50 mM KCl, 10 mM Tris-HCl (pH 8.3), 2.0 mM MgCl2, 0.01% gelatin, 0.2 mM of each deoxynucleoside triphosphate and 0.2 M of
each oligonucleotide primer and stored at 20 C.
The mastermix was thawed and the Amplitaq was added to give
a final concentration of 2.5 U/reaction tube. The reaction mixture
was dispensed into 0.6-ml microcentrifuge tubes and the test sample
was added, either neat or diluted with distilled water, using sterile
50-l glass capillaries. The reaction mixture was overlaid with mineral oil to prevent evaporation. Reaction tubes containing positive
and negative viral DNA control samples were included in each batch
of tests. Nested PCR for CMV, EBV, HSV 1 and VZV was performed
in a DNA thermal cycler (Perkin-Elmer Cetus).
The samples were amplified through 20 cycles consisting of denaturation at 94 C for 1 min, annealing at 60 C for 1 min and primer extension at 72 C for 2 min. Aliquots of 5 l of amplified products were added to 45 l of sterile distilled water and transferred in
a separate room to fresh reaction tubes containing standard mastermix with 2.5 units of Taq polymerase and 1.0 M of each of the nested primers. The reactions were then done according to the standard
protocol for 30 cycles.
Gel electrophoresis of the PCR products
Routine histology
Longitudinally orientated blocks of the full thickness of the bowel wall
were taken for processing into paraffin wax after fixation in 10% formal saline. Sections were cut at a thickness of 6 m and stained with
haematoxylin and eosin (H & E) and periodic acid Schiff (PAS).
Polymerase chain reaction
Paraffin-embedded blocks were cut at a thickness of 10 m and four
sections from each block were dewaxed with xylene, spun in a miTable 1 Details of three patients with idiopathic megarectum and
three with idiopathic megacolon
Idiopathic megarectum Idiopathic megacolon
Pat. 1 Pat. 2 Pat. 3
Pat. 1
Pat. 2 Pat. 3
12
20
42
40
34
37
Duration of
symptoms (years) 10
17
Age at operation
(years)
133
Routine histology
References
Results
PCR
No herpes virus DNA for the four viruses tested was identified in tissue from patients with either idiopathic megarectum or idiopathic megacolon. Positive results were obtained with the control material.
Discussion
Subtle abnormalities of the enteric nervous system are observed in patients with idiopathically dilated large bowel
[37]. We postulated that there may be an underlying viral aetiology in some of these patients, specifically a neurotropic effect of herpes viruses.
PAS staining did not demonstrate any inclusion bodies,
and no herpes virus DNA for EBV, HSV type 1, VZV or
CMV was detected using the PCR technique described.
Springer-Verlag 1998
O R I G I N A L A RT I C L E
135
Introduction
Methods
Double immunohistochemistry
Double labelling with fluorescence and enzymatic detection was
used to assess the coexistence of GAP- and PGP-immunoreactive
nerve fibres. The primary antisera were used at concentrations three
to five times greater for the fluorescence immunolabelling than for
streptavidin-biotin-peroxidase immunohistochemistry due to the
lower sensitivity of the immunofluorescence technique. Two different protocols were used, which proved to be equally sensitive and
specific. Dichlorotriazinyl amino fluorescein (DTAF) and Cy3 were
used as fluorochromes. In protocol 1, the primary mouse antibodywas detected by Cy3-labelled donkey anti-mouse IgG (diluted 1 : 50,
Jackson/Dianova, Hamburg, Germany), and the rabbit primary antisera were detected by DTAF-labelled donkey anti-rabbit IgG (diluted 1 : 25, Jackson/Dianova). In protocol 2, the primary murine antibody was detected by DTAF-labelled donkey anti-mouse IgG (diluted 1 : 25, Jackson/Dianova), and the rabbit primary antiserum by
Cy3-labelled donkey anti-rabbit IgG (diluted 1 : 50, Jackson/Dianova). The tissue sections were incubated with the detection systems
for 2 h at 37 C. Secondary antisera were preabsorbed against serum
proteins of several species including immunoglobulins of the donor
species of the primary antisera to avoid cross reactions. All detection systems were filtered with 0.45-nm disposable filter units (Millipore, Eschborn, Germany). Tests for unspecific binding of the secondary antisera included omission of the primary antibody/antiserum and the detection of the primary antibody/antiserum with noncorresponding detection systems [21]. There was no evidence for
cross reactions. The sections were examined and photographed with
136
a Zeiss axiophot microscope equipped with epi-illumination. To obtain DTAF fluorescence, a 450- to 490-nm filter was used for excitation and a 520-nm filter for the specific emission. A 546-nm filter
was used for Cy3 excitation and a 590-nm filter for the specific emission [2, 21].
Image analysis
The CUE-3 System (Galai Productions, Migdal Haemek, Israel) connected to an Olympus BH-2 microscope via a solid-state video camera (Sony, 3CCD) was used for comparative image analysis, which
was performed on adjacent sections stained for PGP and GAP. In
control sections of uninflamed anal canal, the dermis of the squamous zone and perianal epidermis was analysed because these regions were found to correspond to the main location of the inflammatory process in chronic anal fissures. The system was calibrated
for a 20 objective. All artefacts were excluded from the scanning
area. A threshold was defined for each individual section in order to
distinguish between specific immunostaining and nonspecific background staining. Sections were first scanned for PGP-immunoreactive nerve fibres, followed by an analysis of GAP-immunoreactive
nerve fibres in the same regions on consecutive sections. Hence, the
mean ratio of PGP to GAP staining could be assessed. For the dermis of the normal anal canal, 60 high-power fields were examined
to determine the density of PGP- and GAP-immunoreactive nerve
fibres. To assess the density of PGP- and GAP-immunoreactive innervation of anal fissures to fields were examined under high power. The density of immunoreactive nerve fibres was expressed as
area fraction. To compare the density of PGP- and GAP-immunoreactive innervation, the Wilcoxon rank order test was used for statistical analysis of all measured high-power fields. To compare the
GAP to PGP ratio, the unpaired t-test was used.
Results
immunoreactive nerve fibres, although quantitative differences were obvious in the various regions. In the colorectal zone, GAP-immunoreactive nerve fibres were ubiquitous but less numerous than the PGP-immunopositive innervation. The density of GAP-immunoreactive nerve fibres decreased caudally towards the anal transitional zone,
the squamous zone and perianal epidermis. However, the
density of GAP-immunoreactive innervation of the internal anal sphincter, the musculus canalis ani and the longitudinal muscle was comparable to that of the colorectal
zone. In the dermis of the squamous zone and perianal epidermis, GAP-immunoreactive nerve fibres were contained
in a very minor portion of the pan-neural innervation revealed by PGP (Figs. 1 b, 2 a). In areas of high nerve fibre
density, the GAP-immunopositive innervation constituted
a minor fraction of the PGP-positive nerve fibres.
Changes in PGP- and GAP-immunoreactive innervation
in chronic anal fissures
Histological examination of tissue specimens of chronic
anal fissures revealed that the major inflammatory reaction was located in the dermis of the squamous zone and
perianal epidermis. Portions of the anal transitional zone
were rarely contained in tissue specimens of chronic anal
fissures and were excluded from the analysis. Chronic anal
fissures appeared as ulceration with missing or severely
damaged epidermal layer. All tissue specimens of chronic
anal fissures contained characteristic features of longstanding chronic inflammation, such as fibrosis and infiltrates of lymphocytes.
Changes in PGP- and GAP-immunoreactive innervation were seen in all patients suffering from chronic anal
fissures. In all tissue sections of chronic anal fissures, a
marked increase in the innervation density of PGP- and
GAP-immunoreactive nerve fibres was noted. The hyperinnervation in chronic anal fissures was visible as concentrations of nerve fibre bundles in the dermis (Fig. 3 a, b).
Nerve fibre bundles in chronic anal fissures were enlarged
and in the majority of analysed tissue sections had a neuroma-like appearance (Fig. 2 b). In contrast, the PGP- and
GAP-immunoreactive innervation of the epidermis, if
present in chronic anal fissures, was decreased compared
with control sections. Likewise, the portion of the ulcerations immediately adjacent to the anal canal was not innervated by PGP- or GAP-immunopositive nerve fibres. In
adjacent and identical sections of chronic anal fissures double immunohistochemistry revealed a proportional increase in the GAP-immunoreactive nerve fibre subpopulation compared with the pan-neural PGP-positive nerve
fibres in tissue sections of chronic anal fissures (Fig. 3 a, b).
Quantitative image analysis of PGPand GAP-immunoreactive nerve fibres
Image analysis of adjacent sections alternately stained for
PGP or GAP revealed that the PGP-immunoreactive inner-
137
Fig. 1 Distribution of PGP(A) and GAP- (B) immunoreactive nerve fibres in the dermis
of the uninflamed anal canal revealed by double immunofluorescence. The same section is
shown in A and B. GAP-immunoreactive nerve fibres (arrows
in B) constitute a minor subpopulation of the general innervation (arrows in A), as visualized by staining for PGP. Bar,
50 m
138
termined, which reflects the proportion of GAP-immunoreactive nerve fibres in relation to the pan-neural PGPimmunoreactive area fraction. The mean ratio of GAP to
PGP immunoreactivity was not significantly increased in
chronic anal fissures.
Discussion
In this study, we established the region-specific distribution pattern of GAP-immunoreactive nerve fibres in the
normal anal canal and chronic anal fissures in relation to
the general innervation, as revealed by staining for the panneural marker PGP. In addition, we demonstrated a highly
significant increase in the PGP- and GAP-immunoreactive
innervation in chronic anal fissures as compared with control sections.
The distribution of PGP- and GAP-immunoreactive
nerve fibres in the uninflamed anal canal is in agreement
with previous studies [2, 22, 23]. Interestingly, the proportional distribution of GAP-immunoreactive nerve fibres
was region specific. In compartments innervated by the enteric nervous system, GAP-immunoreactive nerve fibres
constituted a higher proportion of the general innervation
than in compartments innervated by the somatic nervous
system. These differential distribution patterns may indicate elevated plasticity and synaptic remodelling in the enteric nervous system compared with the somatic nervous
system [19, 2224].
In previous studies we reported a very high density of
neuropeptidergic nerve fibres in the dermis of the squamous zone and in the perianal epidermis. This has been
discussed as a possible pathogenic factor for anorectal diseases [2]. In the present study we provide evidence for a
pronounced increase in nerve fibre density in chronic anal
fissures, reflected by an increase in the density of GAPand PGP-immunoreactive nerves. GAP is a member of a
group of brain acid-soluble proteins [2529] and is widely
distributed in the central and peripheral nervous system
[19, 22, 23]. A number of studies has substantiated the
role of GAP in neuronal proliferation and regeneration
[24, 3032]. Recent studies indicate a close association
between elevated levels of GAP in neurons of the peripheral nervous system and chronic inflammation [19, 20]. In
addition, GAP may function as a neuromodulator in the
neuronal release of classical and peptidergic transmitters
[33, 34]. Thus, the pronounced elevation of neuronal GAP
expression in tissue specimens of chronic anal fissures may
not only implicate neuronal proliferation and abortive
sprouting mechanisms in this chronic inflammation, but
also facilitate release of presumed proinflammatory neuronal messengers. Furthermore, the increased nerve fibre
densities observed in anal tissues surrounding chronic fissures might be the result of pathological processes, such
as increased sphincter tone or reduced blood flow [611]
and may thus be secondary in nature.
Nerve fibres in tissue sections of chronic anal fissures
often were neuroma-like in appearance. Neuroma formation occurs during denervation and subsequent loss of neuronal target tissue. Histologically, neuromas are characterized by disrupted perineurium and numerous growth cones.
Hyperalgesia has been found to be associated with neuroma formation [13, 15, 35]. Since neuromas have been
found to have an elevated neuronal discharge [36], neuroma formation in chronic anal fissures may explain the
severe pain that accompanies this chronic inflammation.
Furthermore, alterations in nerve fibre qualities have been
liked to pruritus [37]. In this context it appears likely that
neuroma formation in chronic anal fissures is involved in
the pruritus that accompanies this disorder [6, 9]. Recent
investigations of human chronic pancreatitis lend support
to the view that a possible causal relationship exists
between pain, neuronal plasticity of GAP expression and
immune cell infiltration [38].
Most intriguingly, the putative involvement of the peripheral nervous system in the pathogenesis of anal fissures
suggests new strategies for the therapy of chronic anal fis-
139
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Springer-Verlag 1998
O R I G I N A L A RT I C L E
Abstract The role of paradoxical puborectalis contraction in the aetiology of constipation and how to best diagnose this condition is controversial. The aims of this study
were to investigate whether absolute or relative paradoxical electrical activity during electromyography (EMG) are
related to rectal emptying and to compare EMG, defecography and digital examination in the diagnosis of paradoxical puborectalis contraction. Included in the study were
171 consecutive patients with idiopathic constipation; 136
of these cases were also classified as paradoxical or unclear or not paradoxical at digital examination. Absolute
amplitudes and a strain/squeeze index were used to grade
the EMG activity in the puborectalis and external sphincter muscle. Rectal evacuation was analysed by defecography with image analysis of rectal area. The results showed
that 142 patients had paradoxical EMG activity during
straining. There was a correlation between rectal evacuation and amplitudes (r = 0.20 to 0.03, P < 0.01) and
between evacuation and index (r = 0.34 to 0.39,
P < 0.0001). Forty-two patients with an index of >50 had
impaired rectal evacuation compared with those with an
index 50 (P < 0.0001). Thirty-three of 34 cases (n = 136)
with an index of > 50 also were paradoxical at defecography whereas 19 were diagnosed digitally. In conclusion,
paradoxical puborectalis contraction is associated with impaired rectal evacuation. The activity seems to be best reflected by a strain/squeeze index. The best correlation in
diagnostic methods was between EMG and defecography.
Key words Anismus Constipation Electromyography
Defecography Paradoxical puborectalis contraction
U. Karlbom () W. Graf L. Phlman
Department of Surgery, University Hospital,
S-75185 Uppsala, Sweden
K. Edebol Eeg-Olofsson
Department of Neurophysiology, University Hospital,
Uppsala, Sweden
Introduction
S. Nilsson
Department of Radiology, University Hospital,
Uppsala, Sweden
Paradoxical puborectalis contraction (anismus, spastic pelvic floor syndrome) has been discussed as a pathogenic
142
factor in constipation, whereby a contracting or nonrelaxing puborectalis muscle and external sphincter muscle
would cause an outlet obstruction during attempted rectal
emptying. The paradoxical contraction has been associated
with increased electromyographie (EMG) activity [1] and
anal pressure during straining [2], inability to expel a rectal balloon [3], and prolonged segmental colonic transit
[4]. Failure to increase the anorectal angle [5] or a distinct
impression of the puborectalis muscle during evacuation
have been used as diagnostic criteria on defecography [6].
These criteria have also been associated with impaired rectal evacuation at defecography [5, 6].
Sphincter EMG has been considered a sensitive method
detect paradoxical puborectalis contraction [7] and is used
in many centres in the work-up of severe constipation. In
recent years, biofeedback training has been used most in
the treatment of patients with a paradoxical sphincter response. However, the functional importance of these findings has been questioned, since it has been observed in
other anorectal conditions [8] and in healthy subjects
[9, 10]. It is also questionable whether the laboratory setting is suitable for the evaluation of paradoxical sphincter
contraction. Measurements with ambulatory equipment in
the home environment have led to a reduction in the proportion of patients with paradoxical EMG activity [11].
However, biofeedback training based upon laboratory test
results and directed towards relaxing the sphincter muscles has been successful in several studies [1214] and also
associated with a reduction in sphincter activity during
straining [15]. If paradoxical puborectalis contraction is
clinically relevant, it would affect rectal emptying; however, the relationship between paradoxical EMG activity
and rectal emptying is unclear.
The principal aim of this study was to investigate
whether paradoxical EMG activity is related to rectal evacuation and to evaluate two different grading systems of paradoxical activity. The second aim was to compare EMG,
defecography and digital examination in the diagnosis of
paradoxical puborectalis contraction.
Our routine for defecography have been previously described in detail [6]. Briefly, the anorectal angle was measured as the angle
between the axis of the anal canal and the posterior border of rectum. The size of a rectocele was calculated during straining as the
distance between the maximal anterior outbulge and the axis of the
anal canal. The length of a circular intussusception and the position
of the anorectal junction relative to the ischial tuberosities was measured. Perineal descent was defined as the change in position of the
anorectal junction during straining compared with rest. The defecographic evaluations were performed blindly and X-ray magnification was corrected for in all measurements.
A paradoxical puborectalis contraction was diagnosed when there
was a marked impression of the puborectalis muscle and/or a failure
to increase the anorectal angle during straining compared with rest.
Rectal emptying was evaluated with a computer-based image analysis method [6, 18]. The area with homogeneous contrast in the lower 8 cm of rectum was calculated at rest, after initial evacuation (initial or first straining episode, 0 30 s) and after the total evacuation
period. The time was noted and rectal emptying was expressed as:
(1) percentage evacuated area per second during the initial evacuation; (2) percentage evacuated area per second during the total evacuation period; (3) percentage evacuated area.
143
Digital examination
One hundred and thirty-six of the patients were prospectively assessed with a digital examination according to a protocol. The examination was performed with the patient in the left lateral position.
After identifying the puborectalis and external anal sphincter muscles during squeeze and relaxation, the patient was instructed to strain
as to evacuate. The procedure was repeated at least three times. The
muscle activity during straining was classified as: (1) paradoxical
sphincter contraction; (2) unclear; or (3) not paradoxical. All digital
examinations were performed by three surgeons with a special interest in the field.
Statistical methods
Nonparametrical methods were used. The Kruskall-Wallis test was
used for comparison of several independent groups of patients and
Mann-Whitney U test was used when comparing two groups of patients. Proportions were analysed with Fishers exact test.
Spearmans rank correlation test was used for analysis of correlations.
In a comparison of diagnostic methods, sensitivity was calculated as (true positive/true positive + false negative) 100; specificity
as (true negative/true negative + false positive) 100; positive predictive value as (true positive/true positive + false positive) 100;
and negative predictive value as (true negative/true negative + false
negative) 100. The assumed best method was used to compare
the three diagnostic methods.
Results
Electromyography
Paradoxical activity was found in 142 patients (83%) i.e.
an increment over baseline, during stain. There were 68
patients (39%) with a peak index > 50 and 42 patients (25%)
with a mean index > 50 (Fig. 1). The corresponding amplitude levels, discriminating equal number of patients, were
> 0.9 mV (n = 68) and > 0.76 mV (n = 42). There was an
overall correlation between amplitudes and rectal emptying, but the correlation was stronger between indices and
emptying (Table 1). There was also an inverse relation
Table 1 Overall correlations between EMG activity during straining (amplitude and index) and rectal evacuation measured at defecography in 171 constipated patients
Amplitude (mV)
Rectal evacuation
% Area evacuated
%/s (initial)
%/s (total)
Index
Mean
Peak
Mean
Peak
144
Table 2 Defecographic findings and rectal evacuation according to sphincter muscle activity during straining measured
as mean amplitude or mean index levels at EMG (n = 171)
Anorectal angles
Rest
Strain
Perineal descent (cm)
Mean index
> 0.76
(n = 42)
0.76
(n = 129)
> 50
(n = 42)
50
(n = 129
97 (68 135)
119 (53 150)
95 (44 124) a
92 (28 138) b
2.3 (0 4.8)
2.5 (0 5.7)
2.0 (0 4.9) a
2.7 (0 5.7)
Intussusception (cm)
0 (0 3.6)
0.7 (0 3.9)
0 (0 2.1)
Rectocele (cm)
2.5 (0 3.9)
2.1 (0 5.1)
2.4 (0 4.2)
2.1 (0 5.1)
74 (0 100)
2.5 (0 12.5)
1.3 (0 12.5)
83 (0 100)
4.2 (0 16.6)
2.1 (0 16.6)
47 (0 100) b
1.2 (0 10.3) b
0.6 (0 5.6) b
90 (0 100)
5.2 (0 16.6)
2.7 (0 16.6)
Rectal evacuation
% Area evacuated
%/s (initial)
%/s (total)
0.9 (0 3.6)
Values are median and range; a P < 0.05, b P < 0.001, Mann-Whitney U test
between indices (mean and peak) and length of intussusception (r = 0.23 to 0.22, P< 0.01), perineal descent
(r = 0.25 to 0.22, P< 0.01) and anorectal angle during
straining (r = 0.39 to 0.37, P< 0.001). The anorectal angle during straining was also inversely related to mean amplitude (r = 0.20 P< 0.05), otherwise there were no statistically significant relations between amplitudes and defecographic findings. Those with a mean index > 50 had significantly impaired rectal emptying, less intussusception,
perineal descent and more acute anorectal angles at rest
and at straining, whereas no particular defecographic features were found in those with the highest mean amplitudes
(Table 2). In a corresponding analysis, peak index levels
> 50 related significantly to the same defecographic parameters, whereas peak amplitudes (> 0.9) did not (data not
shown). Expressing the paradoxical puborectalis contraction as index levels rather than amplitudes was more in
agreement with the defecographic diagnosis of paradoxical contraction (proportion positive at defecography: mean
index > 50, 41/42 vs. mean amplitude > 0.76, 19/42;
P < 0.0001, Fishers exact test).
Not paradoxical
(n = 121)
Rectal evacuation
% Evacuated area
%/s (initial)
%/s (total)
48 (0 100)
1.1 (0 7.2)
0.7 (0 2.0)
92 (10 100) a
5.6 (0 16.6) a
2.9 (0.1 16.6) a
EMG
Mean index
Peak index
Mean amplitude
Peak amplitude
56 (4 306)
85 (10 420)
0.7 (0.05 3.7)
1.0 (0.1 6.7)
16 (0 67) a
30 (0 129) a
0.2 (0 5.9) a
0.5 (0 17.8) a
Defecography
A paradoxical puborectalis contraction was diagnosed in
50 patients (29%). The diagnosis was related to a short circular intussusception (P < 0.00001), little perineal descent
(P < 0.008) and an acute anorectal angle at rest (P < 0.002,
data not shown). Paradoxical puborectalis contraction was
also highly related to all rectal evacuation parameters,
EMG amplitudes and EMG indices (Table 3).
Fifty-four patients evacuated all contrast whereas five
patients could not evacuate at all. All patients in the latter
group had a paradoxical puborectalis contraction. Twenty
patients did not evacuate anything during the first 30 s
(initial %/s = 0) and 18 of them were diagnosed as having
a paradoxical contraction at defecography. Of the 25 patients that evacuated less than 0.5%/s during the whole investigation, there were 17 with a paradoxical puborectalis
contraction. In the remaining eight cases the poor empty-
Of 136 patients, 31 (23%) were judged to have a paradoxical puborectalis contraction at rectal examination, and
15 cases (11%) were classified as unclear. The digital examination separated patients with higher indices and
higher amplitudes (Table 4). A digitally diagnosed paradoxical contraction was associated with the same defecographic findings as a paradoxical contraction diagnosed
with EMG indices or with defecography (Fig. 2, Table 4).
Comparison of diagnostic methods
A comparison between diagnostic methods was made
undertaken in the 136 patients who were prospectively assessed by EMG, defecography and digital examination.
The proportions of patients with a paradoxical puborectalis contraction were: defecography 30%, EMG (mean index > 50) 25%, and at digital examination (clearly para-
145
Table 4 Digital classification
in relation to results of EMG
and defecography (n = 136)
Anorectal angles
Rest
Strain
Paradoxical
(n = 31)
Unclear
(n = 15)
Not paradoxical
(n = 90)
Kruskall-Wallis
P value
91 (56 120)
88 (28 150)
0.008
< 0.0001
1.9 (0 4.2)
2.7 (0 5.1)
Intussusception (cm)
0 (0 1.8)
1.2 (0 2.7)
0.6 (0 3.6)
0.0009
Rectocele (cm)
1.8 (0 4.2)
2.4 (0 4.8)
2.3 (0 5.1)
0.58
53 (0 306)
0.7 (0 3.7)
37 (0 94)
0.4 (0 1.2)
17 (0 81)
0.3 (0 5.0)
< 0.0001
0.003
EMG
Mean index
Mean amplitude
0.017
Reference method
Sensitivity
Specificity
Positive
predictive
value
Negative
predictive
value
Defecography a
EMG (mean index > 50)
Digital examination
80
54
99
91
97
71
92
82
97
56
92
88
80
61
99
86
Digital examination a
Defecography
EMG (mean index > 50)
71
61
82
86
54
56
91
88
and 84% (vs defecography) and 68% and 88% (vs EMG),
respectively, if patients with an unclear muscle activity
were included in the paradoxical group. Correspondingly,
specificity and positive predictive value decreased to 80%
and 59% (vs defecography) and to 77% and 50% (vs EMG),
respectively.
Discussion
The high incidence of patients with a paradoxical sphincter activity at EMG during straining and the wide range in
146
A paradoxical puborectalis contraction assessed by digital examination was related to higher indices and also to
higher amplitudes, which makes the digital assessment
clinically relevant. It was also clearly related to impaired
rectal evacuation. It has been suggested that clinical examination is sufficient in most patients with defecatory difficulties [23]. In this study, 15 patients (11%) were difficult
to classify because of varying contractionrelaxation patterns or not fully relaxing muscles during straining. There
were also two patients with weak muscles and were therefore difficult to categorise. However, an unclear diagnosis
might also have some relevance since patients with this
finding had intermediate levels of indices and rectal evacuation.
Although both defecography and EMG are established
methods, none of them can act as a reference method. All
comparisons must be done with the assumption that each
method could be the best. Poor agreement between defecography and EMG has been reported [24, 25]; however,
only a few studies have focused on the methods of diagnosing paradoxical puborectalis contraction. Jorge et al.
[26] found suboptimal correlations between defecography
and EMG when defining the paradoxical contraction as a
failure to achieve a descrease in electrical activity during
attempted defecation. They found a sensitivity and specificity of about 70% and 80%, respectively, for each method.
With the use of a strain/squeeze index (mean index > 50)
as in the present study, the sensitivity and specificity was
improved. Correlations between manometry, EMG and defecography in the diagnosis of paradoxical puborectalis
contraction, which have been studied by Ger et al. [21],
were found to be suboptimal but improved with the use of
a manometric strain/squeeze index.
The agreement between digital examination and
EMG/defecography was not as good as between EMG and
defecography. Embarressment at first visit may explain
some of the false positive results at digital examination.
Siproudis et al. [23] found a negative predictive value of
96% for clinical diagnosis of anismus vs manometric anismus. The corresponding figures in this study were 82%
(vs defecography) and 86% (vs EMG). If unclear diagnoses
were included in the paradoxical group the figures were
84% and 88%, respectively. The results suggest that a finding of normal relaxing sphincter muscles at digital examination can be used to exclude the diagnosis of paradoxical puborectalis contraction in most patients. A positive
diagnosis or an unclear finding requires further evaluation
with other methods.
References
1. Turnbull GK, Lennard-Jones JE, Bartram CI (1986) Failure of
rectal expulsion as a cause of constipation: why fibre and laxatives sometime fail. Lancet: 767769
2. Preston DM, Lennard-Jones JE (1985) Anismus in chronic constipation. Dig Dis Sci 30: 413418
3. Barnes PRH, Lennard-Jones JE (1985) Balloon expulsion from
rectum in constipation of different types. Gut 26: 10491052
147
4. Kuijpers HC, Bleijenberg G, Moree (1986) The spastic pelvic
floor syndrome. Large bowel outlet obstruction caused by pelvic floor dysfunction: a radiological study. Int J Colorectal Dis
1: 4448
5. Papachrysostomou M, Smith AN, Merrick MV (1994) Obstructive defecation and slow transit constipation: the proctographic
parameters. Int J Colorectal Dis 9: 115120
6. Karlbom U, Nilsson S, Phlman L, Graf W (1995) The relationships between defecographic findings, rectal evacuation and colonic tranisit in constipated patients. Gut 36: 907912
7. Wexner SD, Marchetti F, Salanga VD, Corredor C, Jagelman DG
(1991) Neurophysiology assessment of the anal sphincters. Dis
Colon Rectum 34: 606612
8. Jones PN, Lubowski DZ, Swash M, Henry MM (1987) Is paradoxical contraction of puborectalis muscle of functional importance? Dis Colon Rectum 30: 667670
9. Barnes PRH, Lennard-Jones JE (1988) Function of striated anal
sphincter during straining in control subjects and constipated patients with a radiologically normal rectum or idiopathic megacolon. Int J Colorectal Dis 3: 207209
10. Lubowski DZ, King DW, Finlay IG (1992) Electromyography
of the pubococygeus muscles in patients with obstructed defecation. Int J Colorectal Dis 7: 184187
11. Duthie GS, Bartolo DCC (1992) Anismus: the cause of constipation? Results of investigation and treatment. World J Surg
16: 831835
12. Dahl J, Lindquist B, Tysk C, Leissner P, Philipson L, Jrnerot G
(1991) Behavioral medicine treatment in chronic constipation
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34: 769776
13. Wexner SD, Cheape JD, Jorge JMN, Heymen S, Jagelman DG
(1992) Prospective assessment of biofeedback for treatment of
paradoxical puborectalis contraction. Dis Colon Rectum
35: 145150
14. Fleshman JW, Dreznik Z, Meyer K, Fry RD, Carney R, Kodner
IJ (1992) Outpatient protocol for biofeedback therapy of pelvic
floor outlet obstruction. Dis Colon Rectum 34: 17
15. Kawimbe BM, Papachrysostomou M, Binnie NR, Clare N,
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Springer-Verlag 1998
L E T T E R TO T H E E D I TO R
Dear Sir,
Amongst coloproctologists there is
no consensus on the optimal unit to
express anorectal luminal pressures.
American and continental European
workers tend to use millimetres of
mercury whereas their British counterparts prefer centimetres of water.
To illustrate this, at the ECCP meeting in June 1997 nine research groups
(six European, two North American
and one United Kingdom) presented
13 abstracts with anorectal pressures
measured in millimetres of mercury.
In contrast, five other groups presented five abstracts (one European,
four United Kingdom) in centimetres
of water.
Clearly there is a need to adopt
one unit of pressure measurement,
but which is optimal? It is difficult to
be certain, but the likely answer is
that the pascal (Pa) (1 newton/square
metre) is superior to either of the tra-
ditional units currently used. The pascal is the unit of pressure for the
system internationale (SI system).
The SI system is coherent. This means
that any parameter within the system
can be entered into a calculation with
any other parameter within the system
without the need to use mathematical
constants.
Further advantages include the
wide geographical understanding of
the SI system throughout the world
and its increasing utilization within
everyday medicine, for example, in
the expression of blood gas analysis.
Commercially available pressure
manometers are currently calibrated
in traditional units, but it would be
a relatively simple and inexpensive
step to recalibrate in kPa. In order
to field test the use of Pa in the
anorectal physiology laboratory, we
have carried out 30 consecutive
anal pressure measurements expressed in millimetres of mercury and
kPa
1 mmHg = 0.13 kPa .
Springer-Verlag 1998
L E T T E R TO T H E E D I TO R
Dear Sir,
We read with interest the article by
Ho and Tan on ambulant anal manometry before and after hemorrhoidectomy [1]. As mentioned by the
authors, anal pressures are often
raised in patients with piles. It may
be due either to an associated spasm
of the internal anal sphincter (IAS)
or to an increased blood flow and hypertension in the anal cushions forming the piles [2]. The authors state
that, after hemorrhoidectomy without sphincterotomy, there was a significant reduction in the maximum
anal pressures of their patients, consistent with the findings reported by
others [3]. Nevertheless, many surgeons still advocate an internal
sphincterotomy associated with hemorrhoidectomy in patients with
anal spasm evaluated either clinically or manometrically, and some
do it routinely in young adult males
with the aim of reducing postoperative pain.
References
1. Ho YH, Tan M (1997) Ambulatory anorectal manometric findings in patients
before and after haemorrhoidectomy. Int
J Colorect Dis 12:296297
2. Sun WM, Read NW, Shorthouse AJ (1990)
Hypertensive anal cushions as a cause of
the high anal canal pressures in patients
with haemorrhoids Br J Surg 77:458462
3. Ho YH, Seow Choen F, Goh HS (1995)
Haemorrhoidectomy and disordered rectal and anal physiology in patients with
prolapsed haemorrhoids. Br J Surg 82:
596598
4. Favetta U, Amato A, Interisano A, Pescatori M (1996) Clinical, manometric
and sonographic assessment of the anal
sphincters. A comparative prospective
study. Int J Colorect Dis 4:163166