adult ileoileal intussusception induced by an ileal lipoma diagnosed preoperatively: report of a...
TRANSCRIPT
CASE REPORT
Adult ileoileal intussusception induced by an ileal lipomadiagnosed preoperatively: report of a case and reviewof the literature
Tsutomu Namikawa • Norihiro Hokimoto •
Takehiro Okabayashi • Masamitsu Kumon •
Michiya Kobayashi • Kazuhiro Hanazaki
Received: 24 March 2011 / Accepted: 28 June 2011 / Published online: 14 December 2011
� Springer 2011
Abstract We herein report a case of adult ileoileal intus-
susception induced by an ileal lipoma. A 68-year-old woman
with a history of small intestinal tumors was admitted to our
hospital with severe, colicky lower abdominal pain, similar
to episodes experienced in the past. A barium meal enema at
the initial admission demonstrated a small intestinal tumor in
the ileum 30 cm proximal to the ileocecal valve. Abdominal
ultrasound sonography and computed tomography showed a
sausage-shaped mass presenting as a target sign in the right
lower abdomen, suggestive of intussusception. There was
also a round mass of fat attenuation representing a lipoma,
which was considered the lead point of the intussusception.
The patient underwent emergency surgery and partial
resection of the ileum, including the ileal tumor, following
reduction of the intussusception. The resected specimen
contained a round tumor measuring 1.5 9 1.5 9 1.4 cm,
which was diagnosed histopathologically as an intestinal
lipoma. The patient made a satisfactory recovery and was
discharged on postoperative day 10. The clinical character-
istics of previously reported lipomas with intussusception
are also discussed, including the relationships between the
tumor size and symptoms or location.
Keywords Intussusception � Ileal lipoma � Intestinal
tumor
Introduction
Intussusception is the invagination of a bowel loop together
with its mesenteric fold (intussusceptum) into the lumen of a
contiguous portion of bowel (intussuscipiens). Intestinal
intussusception is an infrequent disease that occurs almost
exclusively in children younger than 4 years of age, and is
seldom encountered in adults. Although intussusception
occurs at all ages, there are major differences in the clinical
presentation, diagnostic approach, and management of
intussusception between the pediatric and adult populations
[1]. Moreover, the exact mechanism that precipitates intus-
susception is still unknown. However, it is generally believed
that any lesion in the bowel wall or irritant within the bowel
lumen could alter the normal peristaltic pattern, and thereby
initiate an invagination leading to intussusception [2].
Adult intussusceptions may be caused not only by
benign lesions such as inflammatory polyps, adenomas,
lipomas, neurofibromas, or adhesions, but also by malig-
nant lesions such as small bowel tumors, including primary
cancers and secondary intestinal metastases of other tumors
[1–4]. Despite the evolution of radiological procedures, the
preoperative diagnosis of adult intussusception remains
challenging, particularly with the lack of specific clinical
symptoms and difficulty in examining the small intestine.
The condition is, therefore, often confirmed only at lapa-
rotomy [4, 5].
Gastrointestinal lipomas are rare benign tumors and an
infrequent cause of intestinal intussusception [2, 4].
Ultrasonography (US) and computed tomography (CT) are
useful, both for confirming the existing diagnosis of
T. Namikawa (&) � N. Hokimoto � T. Okabayashi �K. Hanazaki
Department of Surgery, Kochi Medical School, Kohasu,
Oko-cho, Nankoku, Kochi 783-8505, Japan
e-mail: [email protected]
M. Kumon
Department of Surgery, Noichi Central Hospital, Higashino,
Noichi-cho, Konan, Kochi 781-5213, Japan
M. Kobayashi
Department of Human Health and Medical Science,
Kochi Medical School, Kohasu, Oko-cho,
Nankoku, Kochi 783-8505, Japan
123
Surg Today (2012) 42:686–692
DOI 10.1007/s00595-011-0092-6
intussusception and for providing a pathognomonic diag-
nosis of an intestinal tumor as a leading point of the
intussusception [7, 8]. We herein report a case of adult
ileoileal intussusception induced by an ileal lipoma located
close to the ileocecal junction, which was diagnosed by
abdominal US, CT, and barium meal examination. To the
best of our knowledge, this is the sixth case report of adult
ileoileal intussusception caused by an ileal lipoma diag-
nosed preoperatively.
Case report
A 68-year-old Japanese woman was admitted to our hospital
complaining of right hypochondriac pain. A barium meal
examination showed a filling defect in the ileum, measuring
1.5 cm and with clear margins. Abdominal CT was per-
formed to identify the nature of the lesion, and revealed a
1.5 cm fatty lesion in the small intestine (Fig. 1). A col-
onoscopic examination was performed, but could not reach
the location of the lesion. We made a clinical diagnosis of an
ileal tumor and recommended surgical resection. The patient
did not consent to the operation because her symptoms had
dissipated, and she was subsequently discharged.
However, the patient was readmitted to our hospital
3 months later for colicky epigastric pain with associated
nausea and vomiting. A physical examination revealed a
mild distended abdomen without a palpable mass. The lab-
oratory findings on admission were as follows: normal red
blood cell count (442 9 104/mm3; normal range
370–490 9 104/mm3), increased white blood cell count
(10.2 9 103 mm3; normal range 4.0–8.0 9 103/mm3), and
normal platelet count (15.1 9 104/mm3; normal range
14.5–34.0 9 104/mm3). The total protein, alanine amino-
transferase, aspartate aminotransferase, total bilirubin, and
serum creatinine levels were within the normal range, as
were the serum carcinoembryonic antigen and cancer anti-
gen 19-9 levels.
A barium meal examination on her initial admission
showed a round filling defect measuring 1.5 cm with a
clear margin at the terminal ileum portion proximal to the
ileocecal valve (Fig. 2). During the second admission,
ultrasonography showed a multicentric target sign sug-
gestive of an intussusception in the right lower abdominal
region; the mass comprised hyperechoic mesenteric fat
centrally and a peripheral hypoechoic rim (Fig. 3).
Fig. 1 Abdominal computed tomography showing a fatty oval mass
in the small intestine
Fig. 2 The barium meal examination showed a round filling defect
measuring 1.5 cm with a clear margin at the terminal ileum
Fig. 3 Abdominal ultrasonography showed a target sign composed of
central hyperechoic mesenteric fat (arrowhead) with an entering ileal
loop (short arrow) and hypoechoic thickening of the peripheral ileal
wall (long arrow)
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Abdominal CT showed a target sign- or sausage-shaped
lesion typical of an intussusception that varied in appearance
relative to the slice axis (Fig. 4). The inner central area
represented the invaginated intussusceptum, surrounded by
its mesenteric fat and associated vasculature, and all sur-
rounded by the thick-walled intussuscipiens (Fig. 4b–d).
More head-side scans showed a low-density homogenous
mass measuring 1.5 cm that was considered to be the leading
point for the invagination (Fig. 4a). These findings led to a
diagnosis of intussusception induced by lipoma.
Under a clinical diagnosis of small intestinal intussusception
caused by an ileal tumor, which was suspected to be a lipoma,
the patient underwent emergency surgery. The ileum, com-
prising approximately 20 cm of intestine, was invaginated at
the terminal end near the ileocecal valve, and an intussuscep-
tion had occurred (Fig. 5). We gently pushed the intussuscep-
tum back from the distal to the proximal ileum to reduce the
lesion. After manual reduction of the intussusception, it was
palpable as an elastic-soft mass of approximately 1.5 cm in
diameter and not invading the serosa. Approximately 5 cm of
ileum, including the tumor, was resected, and an end-to-end
anastomosis was created. Gross examination of the resected
specimen revealed a round tumor covered with mucosa mea-
suring 1.5 9 1.5 9 1.4 cm (Fig. 6). A microscopic examina-
tion revealed fat cells proliferating in the submucosal layer and
confirmed the diagnosis of ileal lipoma (Fig. 7). The postop-
erative course was uneventful, and the patient was discharged
on postoperative day 10.
Discussion
Intussusception is usually a disease of children aged
between 6 months and 4 years, in which the ileum (intus-
susceptum) telescopes into the colon. Adult intussusception
accounts for only 5–10% of all reported cases [5, 9, 10].
Fig. 4 Abdominal computed tomography showed a small intestinal
intussusception. a A smooth-surfaced, round, low-density mass
measuring 1.5 cm considered to be the leading point was observed
at the distal portion of the invaginated bowel (arrow). b, c A target
sign on a transverse view was seen, comprising a ring-shaped mass
with a thick soft tissue density representing the opposing bowel wall
(arrow) and the central low-density mesenteric fat (arrow head). d A
sausage-shaped mass in the longitudinal view that comprised the
edematous bowel wall (arrow) and mesentery within the lumen
(arrowhead)
688 Surg Today (2012) 42:686–692
123
The classic pediatric symptoms of intussusception, such as
abdominal pain, a palpable mass, and blood in the stool are
rarely found in adults, thus leading to the frequent misdi-
agnosis of this disease in the adult population. Further-
more, unlike pediatric cases, adult intussusception is
usually caused by a tumor, which is commonly located at
or near the ileocecal valve and acts as the lead point for the
intussusception [1, 5, 11]. Many cases in adults are,
therefore, secondary to an underlying lesion, and adult
intussusception of the colon is most often secondary to a
malignant tumor, thereby necessitating surgical interven-
tion [12, 13].
A Medline search was performed using the keywords
‘‘adult intussusception’’ and ‘‘lipoma.’’ Fifty cases of adult
intussusception induced by a lipoma, including our present
case, have been reported in the English literature during the
past decade (Table 1). This cohort comprised 28 men and
22 women with a mean age of 52.1 years. The locations of
the lesions were as follows: 2 cases had lesions in the
stomach, 4 were in the jejunum, 21 were in the ileum, 3
were in the cecum, 7 were in the ascending colon, 6 were in
the transverse colon, 4 were in the descending colon, and in
3 cases, the lesion was located in the sigmoid colon. In the
small intestine, such lesions are most commonly located in
the ileum (84%, 21 of 25 patients), while the jejunum is the
least common site for small intestinal lipomas [14]. The
lipoma size ranged from to 1.2 to 12.0 cm, with a mean
size of 4.9 cm.
Among the 50 cases of adult intussusception induced by
a lipoma, CT was used as the diagnostic modality in 45
patients (90%), endoscopic examinations such as esopha-
gogastroduodenoscopy and colonoscopy was used in 22
(44%), US was used in 16 (32%), and an enema contrast
study was used in 11 patients (22%). Abdominal CT is the
most sensitive radiological method for confirming a diag-
nosis of intussusception [13, 14], with the most frequent
finding on plain abdominal radiographs being a mass of
low-density tissue with or without signs of concomitant
intestinal obstruction [15]. On the other hand, intussus-
ception on US invariably appears as a sausage-shaped mass
consisting of concentric rings when the scan plane is par-
allel to its longitudinal axis, which makes a target sign-like
mass when the scan plane is perpendicular to its longitu-
dinal axis [16, 17].
Such radiological examinations of our patient revealed
the presence of mesenteric fat and its associated
Fig. 5 The ileum was invaginated at the terminal ileum and an
intussusception had occurred (arrow)
Fig. 6 The gross appearance of the resected specimen showed a
polypoid mass with a congested mucosal surface
Fig. 7 A histopathologic examination of the tumor revealed fat cells
proliferating in the submucosal layer
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123
Table 1 The characteristics of the reported cases of adult intussusception induced by a lipoma
Case Author Year Age Gender Diagnostic
modality
Tumor location Tumor size
(cm)
Reference
1 Alkim C 2001 69 Male US, CS Descending colon 4 J Clin Ultrasound
2 Franc-Law JM 2001 42 Male CS, BE, CT, Descending colon 4.5 Am Surg
3 Park KT 2001 39 Male US, CT Ileum 4 J Korean Med Sci
4 Moues CM 2002 72 Male EGD, US, CT Stomach 10 Dig Surg
5 Ahmed HU 2004 28 Male CT Jejunum 3 Ann R Coll Surg Engl
6 Zissin R 2004 20 Female CT Ileum 18 Emerg Radiol
7 Dawes LC 2004 41 Male CT Ileum ND Australas Radiol
8 Triantopoulou C 2004 44 Female CT, CS, ECS Ileum 5 Abdom Imaging
9 Ghidirim G 2005 51 Female US, ECS, CT Cecum 10 Rom J Gastroenterol
10 Jelenc F 2005 56 Male US, CT Ascending colon 6 J Laparoendosc Adv Surg Tech A
11 Adachi S 2005 50 Male ECS, CS, CT Ascending colon 5 Pathol Int
12 Vinces FY 2005 72 Male CT, EGD Stomach 6 Can J Gastroenterol
13 Meshikhes AW 2005 55 Male CT Ileum ND Surg Today
14 Tsushimi T 2006 63 Female US, CT Ileum 2.5 Surg Today
15 Amer NM 2006 73 Female ECS, MRI Sigmoid colon ND Arch Surg
16 McKay R 2006 63 Male CT Ileum 3 JSLS
17 Chiang TH 2006 85 Male US, CT Jejunum 4 J Gastroenterol Hepatol
18 Huh KC 2006 62 Male CT, CS Sigmoid colon 3.5 Dig Dis Sci
19 Abou-Nukta F 2006 55 Female CT Transverse colon 12 Am Surg
20 Croome KP 2007 31 Female CT Ascending colon 5 Can J Surg
21 Atila K 2007 47 Female US, CT Ileum 5 Ulus Travma Acil Cerrahi Derg
22 Atila K 2007 56 Female US, CS, CT Transverse colon 5 Ulus Travma Acil Cerrahi Derg
23 Fatima H 2007 64 Male CS, CT Transverse colon 6 Clin Gastroenterol Hepatol
24 Manouras A 2007 55 Male CT, ECS Jejunum 4 World J Gastroenterol
25 Duijff JW 2007 42 Male US, CT Ileum 3 Case Rep Gastroenterol
26 Lin MW 2007 47 Female CT Ileum 3 J Laparoendosc Adv Surg Tech A
27 Lin I 2008 47 Female CT, CS, Enema Ascending colon 5 Endoscopy
28 Chen R 2008 36 Male CS, CT, ECS Ileum 9 Cases J
29 Akagi I 2008 36 Male CT, ECS Ileum 4 J Nippon Med Sch
30 Wild D 2008 82 Male CS, CT Sigmoid colon 8 Gastrointest Endosc
31 Martin P 2008 69 Male CT, CS Transverse colon 7 Dig Dis Sci
32 Shpaner A 2008 38 Female CS, CT Ileum 3.3 Clin Gastroenterol Hepatol
33 Gurses B 2008 38 Female US, CT, CS Cecum 6 Emerg Radiol
34 Whitfield JD 2009 45 Male CT Ileum 2.5 N Engl J Med
35 Espinel J 2009 43 Female CS, CT Ascending colon 5 Rev Esp Enferm Dig
36 Espinel J 2009 57 Female CS, CT Transverse colon 5.5 Rev Esp Enferm Dig
37 Chung CS 2009 51 Male US, CT, CS Ileum 3 Gastroenterology
38 Dultz LA 2009 77 Male CT Cecum 3.5 JSLS
39 Lin CW 2009 46 Male CS, CT, ECS Descending colon 6 Endoscopy
40 Shiba H 2009 33 Male CT, CS, BE Ileum 4 Case Rep Gastroenterol
41 Walters JB 2009 32 Female CT Ascending colon 5.8 Gastroenterology
42 Laleman W 2009 49 Male US, CT Descending colon 5 Gastroenterology
43 Paskauskas S 2010 53 Female US, CS, ECS Ascending colon 7 Medicina (Kaunas)
44 Chuang MT 2010 26 Female CT Ileum ND Am J Surg
45 Mason R 2010 51 Female CT Transverse colon 6.2 J Gastroenterol Hepatol
46 Wan XY 2010 68 Male CS Jejunum 3.2 World J Gastroenterol
47 Abbasakoor NO 2010 52 Female CT Ileum 3.2 J Med Case Reports
48 Kuzmich S 2010 62 Female US Ileum 7 J Clin Ultrasound
690 Surg Today (2012) 42:686–692
123
vasculature inside a dilated loop. Furthermore, a homoge-
neous intraluminal mass consistent with fat in the intus-
susception raised the possibility of a lipoma. Accordingly,
CT and US are valuable methods not only for diagnosing
the presence of an intussusception, but also for revealing
the lesion as the leading point or cause of the intussus-
ception. A preoperative diagnosis of intussusception was
made in 21 (42%) patients in our review. Almost all of
these lipomas presented with characteristic fatty densito-
metric values and a smooth margin, and were consequently
well demonstrated by CT or US, which was frequently
pathognomonic.
Previous reports have suggested that 52–80% of adult
cases of small intestinal intussusception are caused by
benign entities such as lipomas, hamartomatous polyps,
inflammatory polyps, hyperplastic polyps, Meckel’s
diverticulum, and surgery-related lesions or adhesions [4,
12]. Indeed, small intestinal tumors are rare, accounting for
only 1–2% of all gastrointestinal tract tumors [6]. Lipomas
of the small intestine are rare benign tumors with no
malignant potential and are most commonly encountered
incidentally, since they are usually asymptomatic [18]. In
the present case, although the initial examination revealed
a fatty, dense mass with clear margins, suggestive of
lipoma, we recommended surgical resection because of the
symptomatic tumor.
In our review of the cases of lipomas with intussus-
ception, abdominal pain that was often colicky and inter-
mittent was the most common clinical presentation (84%,
42 of 50 patients). Other common symptoms were intesti-
nal obstructive syndrome, such as nausea and vomiting,
gastrointestinal bleeding due to ulceration of the overlying
mucosa, and an abdominal palpable mass. However, there
were no significant differences in the tumor size between
patients presenting with and without abdominal pain
(4.9 ± 2.4 vs. 4.9 ± 2.3 cm, P = 0.998). Therefore, it
seems that most lipomas associated with intussusception
give rise to abdominal pain, irrespective of the tumor size.
When the previously reported 50 cases were divided into
two groups according to the tumor location, the tumor size
was significantly greater in the colon group than in the
patients with an upper gastrointestinal tract location,
including stomach and small intestine (6.0 ± 2.0 vs.
4.0 ± 2.1 cm, P = 0.003) (Table 2). The luminal diameter
of the small intestine is smaller than that of the colon, thus,
smaller tumors might result in intussusception more fre-
quently then larger colonic tumors.
Definitive surgical resection remains the recommended
treatment for adult intussusception due to the large pro-
portion of structural causes and the relatively high inci-
dence of malignancy; however, the optimal surgical
management remains controversial [12, 13, 19]. The high
proportion of malignant lesions in adult cases of intussus-
ception argues for resection without reduction procedures,
such as preoperative barium or air. However, in the small
intestine, malignancy is less frequently associated with
intussusception, even though neoplasms remain the leading
causative lesion [11]. Some investigators have stated that
Table 1 continued
Case Author Year Age Gender Diagnostic
modality
Tumor location Tumor size
(cm)
Reference
49 Balamoun H 2011 65 Male CT Ileum 1.2 World J Gastrointest Surg
50 Our case 2011 68 Female US, CT, ECS Ileum 1.5 Surg Today
CT computed tomography, MRI magnetic resonance imaging, CS colonoscopy, ECS enema contrast study, EGD esophagogastroduodenoscopy,
US ultrasonography, ND not described
Table 2 The characteristics of adult intussusception induced by a
lipoma according to the tumor location
Stomach and
small
intestine
Colon P value
Age in years (mean ± SD) 50.1 ± 16.1 54.5 ± 13.5 0.302
Gender 0.283
Male 17 11
Female 10 12
Tumor location
Stomach 2 0
Jejunum 4 0
Ileum 21 0
Cecum 0 3
Ascending colon 0 7
Transverse colon 0 6
Descending colon 0 4
Sigmoid colon 0 3
Tumor size in cm
(mean ± SD)
4.0 ± 2.1 6.0 ± 2.0 0.003
Diagnosis 0.704
Preoperative 12 9
Postoperative 15 14
Surg Today (2012) 42:686–692 691
123
small bowel intussusception should still be reduced only in
patients in whom a definitive benign diagnosis has been
made preoperatively, or in patients in whom resection may
result in short gut syndrome [13, 19]. However, without
reduction, the surgical resection of a long segment of
intussusception often necessitates the excision of more of
the intestine than necessary to remove the tumor, because it
might also compromise the mesenteric vessels. Therefore,
reduction could be attempted if the involved bowel seg-
ment is viable and when a diagnosis of a benign lesion has
been reached preoperatively. Such an approach results in
less invasive surgery for the intussusception and may thus
allow surgeons to avoid an overly extensive resection of
the intestine.
The present case highlights the possibility of intussus-
ception with an unusually benign cause, such as lipoma,
when adult patients present with nonspecific abdominal
symptoms and small bowel obstruction. Additional reports
of intussusception in the adult population are needed to
optimize the standard management for this uncommon
disease.
Conflict of interest Tsutomu Namikawa and other co-authors
declare no conflicts of interest.
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