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Ileosigmoid knotting: a report of two cases
N. J. Jebbin
Department of Surgery, University of Port Harcourt Teaching Hospital, Port Harcourt, Nigeria
Correspondence to: Dr N. J. Jebbin (E-mail: [email protected])
Abstract
Background: Ileosigmoid knotting (ISK) is a rare
cause of acute intestinal obstruction with high
morbidity and mortality. The diagnosis is rarely
made preoperatively because of its infrequency and
the variations in presentation.
Aim: To report two cases managed by the author,
which will hopefully increase awareness of this
condition.
Case Reports: The first was a 51-year-old man who
presented with a sudden onset of colicky lower
abdominal pain, which later became generalized.
His pulse rate was 100 beats/minute while his blood
pressure was 80/60 mmHg.There was mild
abdominal distension with absent bowel sounds.
The second was a 50-year-old man with a sudden
onset of generalized colicky abdominal pain,
abdominal distension and vomiting. His pulse rate
was 120 beats/minute and the blood pressure 80/50
mmHg. His bowel sounds were markedly reduced.
Introduction
Ileosigmoid knotting is an unusual and rare cause of
intestinal obstruction. It is more commonly seen in
parts of Africa, Asia, and the Middle East but less so
in other parts of the world like the UK where
1, 2
reports are sporadic . Parker first described the
condition in 1845; since then, over 260 cases have
been reported from different parts of the world, but
2
the exact incidence is not known . It occurs
commonly in men in their 4th decade but has been
reported in children also, including a neonate3, 4. This
is a report of two cases of ISK managed by the
author in Mile One Hospital, Port Harcourt,
Nigeria. The literature was reviewed.
Port Harcourt Medical Journal 2007; 1: 197-200
They were resuscitated accordingly. In both
patients, exploratory laparotomy revealed a copious
amount of sero-sanguinous fluid in the peritoneal
cavity with ileosigmoid knotting, and extensive
gangrene involving the distal ileum and the sigmoid
colon. In the first patient, the caecum was involved
in the knot and therefore also gangrenous. Each of
them had a sigmoid colectomy with a right
hemicocetomy. The first patient died of adult
respiratory distress syndrome while the other had an
uneventful recovery. In the patient who died,
surgery was done on the third day of onset of
symptoms.
Conclusion: A high index of suspicion, aggressive
resuscitation, and prompt surgical intervention are
indispensable for a successful outcome.
Key Words: Ileosigmoid knotting, Gangrene, Right
hemicolectomy, Sigmoid colectomy
Case reports
Case 1
A 51-year-old man presented with a sudden onset of
colicky lower abdominal pain, which later became
generalized. The pain was associated with vomiting
of recently ingested food and constipation. He
visited a private clinic, where he was treated for
about ten hours with no improvement. He therefore
went to Mile One Hospital, Port Harcourt for reevaluation. Physical examination revealed an ill
looking, pale middle-aged man in mild respiratory
and painful distress. His pulse rate was100
beats/minute, regular with moderate volume. His
blood pressure was 80/60 mmHg. The abdomen
was mildly distended with generalized tenderness.
197
Ileosigmoid knotting: a report of two cases
The bowel sounds were hardly audible. A
provisional diagnosis of acute intestinal obstruction
was made. He was then resuscitated with
cr ystalloids, broad-spectr um antibiotics
(ceftriazone, gentamycin and metronidazole), and a
naso-gastric tube was passed. Plain erect/supine
radiographs of the abdomen revealed distended
loops of small and large bowel with multiple
air/fluid levels.
An exploratory laparotomy carried out the
following day revealed a copious sero-sanguinous
fluid and an ileo-sigmoid knotting with gangrene
involving the sigmoid colon, distal third of the
ileum and caecum (Figure1). A sigmoid colectomy
with primary colo-colic anastomosis and extended
right hemicolectomy was perfor med.
Unfortunately, the patient died three days later from
adult respiratory distress syndrome.
N. J. Jebbin
80/50mmHg. The abdomen was mildly distended
with generalized tenderness and hyperactive bowel
sounds. A diagnosis of acute intestinal obstruction
was made and he was resuscitated with crystalloids,
nasogastric suction and broad spectrum antibiotics
(ceftriazone, gentamycin and metronidazole). An
exploratory laparotomy performed the same day
revealed a copious amount of serosanguinous fluid
with ileosigmoid knotting and extensive gangrene
involving the distal half of the ileum and sigmoid
colon. An extended right hemicolectomy with ileocolic anastomosis and sigmoid colectomy with
primary colo-colic anastomosis were done. His
postoperative period was uneventful except for a
few bouts of diarrhoea and he was discharged 10
days after surgery. He was lost to follow-up after two
visits.
Discussion
Figure 1. Gangrenous bowel
Case 2
A 50-year-old man presented with a sudden onset
of generalized colicky abdominal pain, abdominal
distension, and vomiting of six hours duration. On
examination, he was covered with sweat, pale and
dehydrated. His pulse rate was 120 beats/minute
with poor volume. The blood pressure was
Port Harcourt Medical Journal 2007; 1: 197-200
Ileosigmoid knotting is a very rare and curious cause
of intestinal obstruction, occurring in men in their
4th decade3. It is relatively uncommon in females5. It
is also referred to as compound volvulus or double
volvulus but recently Raveenthiran has suggested
that the correct terminology should be compound
2
volvulus .
The aetiology of ISK is not very clear but
anatomic and dietary factors have mainly been
incriminated. These include a long small intestinal
mesentery with a freely mobile small bowel, a long
sigmoid loop on a narrow pedicle, and the
consumption of a high bulk diet in the presence of
an empty small bowel. Meckel's diverticulum has
6
been reported to be present in 14-53% of cases
while a relaxed anterior abdominal wall, as
commonly seen in postpartum women has also
7
been implicated .
Shepherd's description of the pathogenesis of
3
ISK has been universally accepted . In his
description, loops of ileum descend into the left
paracolic gutter and wrap around the sigmoid colon,
both becoming distended sooner or later. With time,
both loops of ileum and sigmoid become
strangulated, the latter more frequently so.
Gangrene ensues eventually, and may extend to the
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Ileosigmoid knotting: a report of two cases
terminal ileum, occasionally involving the caecum
and rarely, the proximal ascending colon1, 3.
The diagnosis of ISK is infrequently made
preoperatively, this being possible only in about
1, 7
20% of patients . This is because the symptoms are
often nonspecific. Any patient previously in a good
state of health and developing a sudden onset of
colicky abdominal pain, other features of intestinal
obstruction, and in a shock state, all occurring in
3,8
rapid succession should arouse a suspicion of ISK .
The two cases in this report presented in this
manner.
White blood cell count usually shows
9
leucocytosis due to gangrene and it is thus not
specific for ISK. Plain abdominal radiographs may
show characteristic double closed loop obstruction,
with the sigmoid on the right upper quadrant and
the small bowel loops on the left but this is only an
1,7
occasional finding . More often, the picture is
either that of simple sigmoid volvulus or small
bowel obstruction but the former is predominant.
Computerized tomographic features of ISK have
been described. These include the 'whirl sign',
created by the twisted intestine and mesentery, and
medial deviation of the descending colon with a
beak appearance on its medial border10. This
investigation was not available in Port Harcourt at
the time.
Survival in this rare but serious malady hinges
on aggressive resuscitation and early surgical
intervention 1,11 . The diagnosis of intestinal
obstruction without a definite pathology is a
sufficient indication for a laparotomy. It has been
observed that accurate diagnosis is not an end in
itself but a means to providing an appropriate
treatment12. The surgical option is dictated by the
findings at laparotomy. With viable intestinal loops,
untying the knot alone is considered sufficient, and
recurrence is uncommon3 . Other authors advocate
resection of the sigmoid colon in all cases13 or
mesosigmoidoplasty11 to avert recurrence. When
the bowel loops are gangrenous, most authors
recommend the knot should not be unraveled as this
can lead to bowel perforation and septic shock 1,3.
Gangrene of both loops of bowel will demand en
block resection of the knot and double primary
Port Harcourt Medical Journal 2007; 1: 197-200
N. J. Jebbin
anastomosis of ileal and sigmoid ends3,7. However,
if gangrene extends to less than 10cm from the
ileocaecal junction, a right hemicolectomy is
recommended1. This was the option adopted in the
cases presented. Other options include closure of
the distal ileal stump and end to side ileo-colic
anastomosis 3, 7. The disadvantage of this is that the
bypassed portion of the intestine, known as a blind
loop, often initiates a cascade of problems known as
14
the blind loop syndrome . Thus it is advised when
the patient's intra operative condition is precarious.
The prognosis of ISK depends on early
2,8
diagnosis and eventual surgery . The mortality
rates of non-gangrenous ISK range from 6.8%-8%.
For gangrenous cases, however, they vary from
20%-100%15. Septic shock is the commonest cause
of death13. The first patient presented died from
ARDS following septic shock. Poor prognostic
factors include age over 60 years, duration of
symptoms longer than 24 hours, and extensive
bowel gangrene1.
Conclusion
Ileo-sigmoid knotting is a rare cause of intestinal
obstruction. Its diagnosis often tasks even the
clinical acumen of experienced surgeons. This is
therefore very often made at laparotomy. The
prognosis used to be very poor but currently, with
early diagnosis, aggressive resuscitation, potent
broad-spectrum antibiotics and advances in
anaesthesia with modern post-operative intensive
care, the outcome is improving.
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