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Adult intussusceptions caused by a lipoma in the jejunum: report of a case and review of the literature.
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PMID:  22913731     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Intussusceptions in adults is rare. Gastrointestinal lipomas are rare benign tumors and intussusceptions due to a gastrointestinal lipoma constitutes an infrequent clinical entity. Lipoma may develop as a benign tumor in all organs and rarely in large or small intestine. The present report describes a case of jejunojejunal intussusceptions in an adult with a history of colicky upper abdominal pain. Ileo-ileal invagination was diagnosed by computed tomography scan. Exploratory laparotomy revealed jejunojejunal intussusceptions secondary to a lipoma which was successfully treated with segmental intestinal resection. A review of the literature is also performed regarding this rare association revealing the diagnostic and therapeutic debates that exist. ABSTRACT (FRENCH): L'invagination chez les adultes est rare. Les lipomes gastro-intestinaux sont de rares tumeurs bénignes et l'invagination intestinale due à un lipome gastro-intestinal constitue une entité clinique trés rare. Le lipome peut se développer comme une tumeur bénigne dans tous les organes et rarement dans l'intestin grêle ou le colon. Le présent rapport décrit un cas d'invagination jéjunojéjunale chez un adulte avec une histoire de douleurs abdominales. Iléo-iléale invagination a été diagnostiquée par tomodensitométrie. Une laparotomie exploratrice a révélé l'existence d'une invagination jéjunojéjunale secondaire à un lipome qui a été traitée avec succès par une résection intestinale segmentaire. Une revue de la littérature est également effectuée au sujet de cette association rare révélant les débats diagnostiques et thérapeutiques qui existent.
Authors:
Ouadii Mouaqit; Hafid Hasnai; Leila Chbani; Bachir Benjelloun; Hicham El Bouhaddouti; Karim Ibn El Majdoub; Imane Toughrai; Said Ait Laalim; Abdelmalek Oussaden; Khalid Maazaz; Afaf Amarti; Khalid Ait Taleb
Publication Detail:
Type:  Journal Article     Date:  2012-08-22
Journal Detail:
Title:  World journal of emergency surgery : WJES     Volume:  7     ISSN:  1749-7922     ISO Abbreviation:  World J Emerg Surg     Publication Date:  2012  
Date Detail:
Created Date:  2012-11-20     Completed Date:  2012-11-21     Revised Date:  2013-03-21    
Medline Journal Info:
Nlm Unique ID:  101266603     Medline TA:  World J Emerg Surg     Country:  England    
Other Details:
Languages:  eng     Pagination:  28     Citation Subset:  -    
Affiliation:
Surgery Department, University Hospital Hassan II, Fez 30000, Morocco. mouaqit1975@hotmail.com.
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Journal ID (nlm-ta): World J Emerg Surg
Journal ID (iso-abbrev): World J Emerg Surg
ISSN: 1749-7922
Publisher: BioMed Central
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Copyright ©2012 Mouaqit et al.; licensee BioMed Central Ltd.
open-access:
Received Day: 12 Month: 4 Year: 2012
Accepted Day: 16 Month: 8 Year: 2012
collection publication date: Year: 2012
Electronic publication date: Day: 22 Month: 8 Year: 2012
Volume: 7First Page: 28 Last Page: 28
PubMed Id: 22913731
ID: 3502287
Publisher Id: 1749-7922-7-28
DOI: 10.1186/1749-7922-7-28

Adult intussusceptions caused by a lipoma in the jejunum: report of a case and review of the literature
Ouadii Mouaqit1 Email: mouaqit1975@hotmail.com
Hafid Hasnai1 Email: hasnai_hafid@hotmail.com
Leila Chbani2 Email: chbanil@yahoo.fr
Bachir Benjelloun1 Email: benjelloun19@gmail.com
Hicham El Bouhaddouti1 Email: h.elbouhaddouti@yahoo.fr
Karim Ibn el Majdoub1 Email: ibnmajdoubkarim@gmail.com
Imane Toughrai1 Email: toughiman@hotmail.com
Said Ait Laalim1 Email: aitlaalims@yahoo.fr
Abdelmalek Oussaden1 Email: aousadden@yahoo.fr
Khalid Maazaz1 Email: khali.mazaz@gmail.com
Afaf Amarti2 Email: amartiafaf@yahoo.fr
Khalid Ait Taleb1 Email: khaittaleb@gmail.com
1Surgery Department, University Hospital Hassan II, Fez 30000, Morocco
2Department of pathology, University Hospital Hassan II, Fez, 30000, Morocco

Background

Intussusceptions was reported for the first time in 1674 by Barbette of Amsterdam [1]. The occurrence of intussusceptions in adults is rare, accounting for less than 5% of all cases of intussusceptions and almost 1%-5% of bowel obstruction [2]. In contrast to pediatric intussusceptions, which is idiopathic in 90% of cases, adult intussusceptions has an organic lesion in 70% to 90% of cases [3]. The majority of lipomas in the small bowel are solitary. Approximately 5% are multiple [4]. Symptomatic lipoma manifestations are hemorrhage or intestinal obstruction. Due to their intramural location, lipomas can also serve as the leading point for intussusceptions. We report a rare case of jejuno-jejunal intussusceptions in an adult secondary to an jejunal lipoma.


Case presentation

A 35-year-old man was admitted to the emergency department in a tertiary referral hospital with 4 months history of intermittent upper abdominal pain accompanied with nausea. The patient had no past history of peptic ulcer disease, alteration in bowel habits, melena or weight loss. On examination, he was apyrexial and hemodynamically stable. His abdomen was distended and no palpable abdominal masses; bowel sounds were hyper audible. Initial A rectal examination revealed no masses or blood. Laboratory blood tests were normal. Abdominal radiography revealed prominent dilatation of the small bowel with air fluid levels (Figure  1). Abdominal CT showed a target sign- or sausage-shaped lesion typical of an intussusceptions that varied in appearance relative to the slice axis (Figure  2). The inner central area represented the invigilated intussuscepted, surrounded by its mesenteric fat and associated vasculature, and all surrounded by the thick-walled intussuscipiens. More head-side scans showed a low-density homogenous mass measuring 4 cm that was considered to be the leading point for the invagination (Figure  3). These findings led to a diagnosis of intussusceptions induced by a tumor most likely begin. The decision was made to undertake an urgent exploratory laparotomy. At laparotomy, 50 cm distal to the ligament of Treitz, a jejuno-jejunal intussusceptions was identified. We conducted a desinvagination Benin saw the character of the lesion on CT. The presence of irreversible ischemia in a small portion of the intussusceptum necessitated segmental resection and primary anastomosis (Figure  4). The postoperative period was uneventful and the patient was discharged on the sixth postoperative day. Gross examination of the respected specimen revealed a round tumor covered with mucosa measuring 6 cm. A microscopic examination revealed fat cells proliferating in the submucosal layer and confirmed the diagnosis of ileal lipoma (Figure  5). The histopathology report confirmed a 60-mm submucosal lipoma in the jejunum as a cause for a 30-cm jejuno-jejunal intussusceptions. There was no evidence of dysplasia or malignancy.


Discussion

Intussusceptions in adulthood is unusual, with an incidence of approximately 2-3 cases per population of 1 000 000 per year [5]. The most common classification system divides intussusceptions into four categories: enteric, ileocolic, ileocaecal and colonic [1-4]. In adults, intussusceptions is more likely to present insidiously with vague abdominal symptoms and rarely presents with the classic triad of vomiting, abdominal pain and passage of blood per rectum, making diagnosis difficult [6]. Tumors of the small bowel account for only 1% to 2% of all gastrointestinal tumors, and benign tumors account for approximately 30% of all small-bowel tumors [7]. Lipomas are benign tumors of mesenchymal origin. They are the second most common benign tumors in the small intestine and account for 10% of all benign gastrointestinal tumors and 5% of all gastrointestinal tumors [1,2,5]. Gastrointestinal lipomas are most commonly located in the colon (65% to 75%), small bowel (20% to 25%) and occasionally in the foregut (< 5%) [8]. Fifty-one cases of adult intussusceptions induced by a lipoma, including our present case, have been reported in the English literature during the past decade (Table  1) [9]. Lipomas are largely asymptomatic. The majority of presenting features are either intestinal obstruction or hemorrhage [1,2,5-8]. Their usual location in the small intestine is ileum (50%) while jejunum is the least common. The peak age of incidence is in the 6th-7th decades of life and it appears that females are more prone to lipomas. Malignant degeneration has never been reported [5]. The clinical presentation is very non-specific which makes this a difficult condition to diagnose. According to the literature, only 32% to 50% of cases are diagnosed preoperatively, despite the evolution of imaging methods [9-11]. Abdominal pain, nausea, diarrhea and bleeding per rectum are the common symptoms. Rarely, this can present with acute intestinal obstruction. The classical triad of abdominal pain, sausage shaped palpable mass and passage of red current jelly stools seen in children is rarely seen in adults. Fewer than 20% of cases present acutely with complete bowel obstruction. A palpable abdominal mass is present in only 7% to 42% of cases [12,13]. Lipomas can be diagnosed through conventional endoscopy, capsule endoscopy, barium studies and, most importantly, CT scan [14]. Ultrasound is usually the first modality to be recruited. However, it is operator-dependent and the presence of distended bowel decreases the ability to demonstrate the site of the obstruction. Computed tomography is the imaging method of choice for diagnosing intussusceptions. A submucosal lipoma can be diagnosed if a smooth well-circumscribed mass of fat density (-50 to -100 Hounsfield Units) is revealed within the lumen of the bowel or intussuscipiens. The sensitivity of CT scan to correctly diagnose intussusceptions has been reported from 71.4%-87.5% while its specificity in adults has been reported to be 100% as verified by the subsequent surgery [14,15]. Capsule endoscopy and digital balloon endoscopy are newer means for diagnosing lipomas and are particularly helpful in cases involving small bowel lipomas [8]. Definitive surgical resection remains the recommended treatment for adult intussusceptions due to the large proportion of structural causes and the relatively high incidence of malignancy; however, the optimal surgical management remains controversial [1,2,6,7,9]. Some investigators have stated that small bowel intussusceptions 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 [9]. The dangers of transperitoneal, vascular, and intraluminal seeding after exposing and handling friable and edematous malignant tissues has led many surgeons to advocate en bloc resection of the lesion. All surgeons agree, though, that reduction should not be attempted if there are signs of irreversible bowel ischemia, inflammation or when malignancy is being suspected [5,16,17]. The advantages of intraoperative reduction of the intussusceptions prior to resection, especially when the small bowel is affected, are that it may preserve a considerable length of bowel and thereby prevent development of short-bowel syndrome.


Conclusion

The lipoma is a rare benign tumor of the digestive tract. The diagnosis of intussusceptions in adults can be difficult because of atypical and episodic symptoms. A high level of clinical suspicion and an abdominal CT scan are most useful tools for making a timely diagnosis. Surgical resection remains the treatment of choice and produces an excellent prognosis.


Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images


Abbreviations

CT: Computed tomography; MRI: Magnetic resonance imaging; CS: Colonoscopy; ECS: Enema contrast study; EGD: Esophagogastroduodenoscopy; US: Ultrasonography; ND: Not described.


Competing interests

The authors declare that they have no competing interests.


Authors’ contributions

All of the authors were involved in the preparation of this manuscript. OM performed the operation and revised the manuscript. HH was an assistant surgeon and made substantial contributions to conception and design. LC described histological finding and was involved in drafting the manuscript. All authors read and approved the final manuscript.


References
Krasniqi AS,Hamza AR,Salihu LM,Spahija GS,Bicaj BX,Krasniqi SA,et al. Compound double ileoileal and ileocecocolic intussusception caused by lipoma of the ileum in an adult patient: A case reportJ Med Case ReportsYear: 20115145210.1186/1752-1947-5-452
Balamoun H,Doughan S,Ileal lipoma. A rare cause of ileocolic intussusception in adults: Case report and literature reviewWorld J Gastrointest SurgYear: 201131131510.4240/wjgs.v3.i1.1321286220
Balik AA,Ozturk G,Aydinli B,Alper F,Gumus H,Yildirgan MI,et al. Intussusception in adultsActa Chir BelgYear: 200610640941217017694
Atila K,Terzi C,Obuz F,Yilmaz T,Füzün M,Symptomatic intestinal lipomas requiring surgical interventions secondary to ileal intussusception and colonic obstruction: report of two casesUlus Travma Acil Cerrahi DergYear: 20071322723117978899
Andreas M,Lagoudianakis EE,Dimitrios D,Tsekouras DK,Markogiannakis H,Genetzakis M,et al. Lipoma induced jejunojejunal intussusceptionsWorld J GastroenterolYear: 200713263641364417659719
Ali A,Morteza N,Rasoul M,Bodaghabadi M,Mardany O,Ali FA,et al. Ileal intussusception secondary to both lipoma and angiolipomaA case report Cases JYear: 200927099
Akagi I,Miyashita M,Hashimoto M,Makino H,Nomura T,Tajiri T,Adult intussusception caused by an intestinal lipoma: report of a caseJ Nihon Med SchYear: 200875316617010.1272/jnms.75.166
Chou JW,Feng CL,Lai HC,Tsai CC,Chen SH,Hsu CH,et al. Obscure gastrointestinal bleeding caused by small bowel lipomaIntern MedYear: 2008471601160310.2169/internalmedicine.47.096318797119
Namikawa T,Hokimoto N,Okabayashi T,Kumon M,Kobayashi M,Hanazaki K,Adult ileoileal intussusception induced by an ileal lipoma diagnosed preoperatively: report of a case and review of the literatureSurg TodayYear: 201242768669210.1007/s00595-011-0092-622167483
Barussaud M,Regenet N,Briennon X,de Kerviler B,Pessaux P,Kohneh-Sharhi N,et al. Clinical spectrum and surgical approach of adult intussusceptions: a multicentric studyInt J Colorectal DisYear: 20062183483910.1007/s00384-005-0789-315951987
Haas EM,Etter EL,Ellis S,Taylor TV,Adult intussusceptionAm J SurgYear: 20031861757610.1016/S0002-9610(03)00108-912842754
Thompson WM,Imaging and findings of lipomas of the gastrointestinal tractAJR Am J RoentgenolYear: 20051841163117115788588
Huang BY,Warshauer DM,Adult intussusception: diagnosis and clinical relevanceRadiol Clin North AmYear: 2003411137115110.1016/S0033-8389(03)00116-714661662
Kuzmich S,Connelly JP,Howlett DC,Kuzmich T,Basit R,Doctor C,Ileocolocolic intussusception secondary to a submucosal lipoma: an unusual cause of intermittent abdominal pain in a 62-year-old womanJ Clin UltrasoundYear: 2010381485119655322
Barbiera F,Cusma S,Di Giacomo D,et al. Adult intestinal intussusception: comparison between CT features and surgical findingsRadiol Med (Torino)Year: 2001102374211677436
Hadithi M,Heine GD,Jacobs MA,van Bodegraven AA,Mulder CJ,A prospective study comparing video capsule endoscopy with double-balloon enteroscopy in patients with obscure gastrointestinal bleedingAm J GastroenterolYear: 2006101525710.1111/j.1572-0241.2005.00346.x16405533
Chiang TH,Chang CY,Huang KW,Liou JM,Lin JT,Wang HP,Jejunojejunal intussusception secondary to a jejuna lipoma in an adultJ Gastroenterol HepatolYear: 20062192492610.1111/j.1440-1746.2006.04123.x16704553

Figures

[Figure ID: F1]
Figure 1 

Abdominal X-Ray. In favor of bowel obstruction.



[Figure ID: F2]
Figure 2 

Abdominal computed tomography. Showing a fatty oval mass in the small intestine.



[Figure ID: F3]
Figure 3 

Computed tomography scan of the abdomen without oral contrast. A longitudinal cut view of the intussusception shows the “sausage” shape.



[Figure ID: F4]
Figure 4 

Intraoperative findings of the lipoma: A pedunculated lesion, measuring 60 mm, was the lead point of the intussusception.



[Figure ID: F5]
Figure 5 

Histological findings of the tumor. A histopathologic examination of the tumor revealed fat cells proliferating in the submucosal layer.



Tables
[TableWrap ID: T1] Table 1 

The characteristics of the reported cases of adult intussusception induced by a lipoma


Case Age Gender Diagnostic modality Tumor location Size (cm) Reference
1
69
Male
US, CS
Descending colon
4
J Clin Ultrasound
2
42
Male
CS, BE, CT
Descending colon
4.5
Am Surg
3
39
Male
US, CT
Ileum
4
J Korean Med Sci
4
72
Male
EGD, US, CT
Stomach
10
Dig Surg
5
28
Male
CT
Jejunum
3
Ann R Coll Surg Engl
6
20
Female
CT
Ileum
18
Emerg Radiol
7
41
Male
CT
Ileum
ND
Australas Radiol
8
44
Female
CT, CS, ECS
Ileum
5
Abdom Imaging
9
51
Female
US, ECS, CT
Cecum
10
Rom J Gastroenterol
10
56
Male
US, CT
Ascending colon
6
J Laparoendosc Adv Surg Tech A
11
50
Male
ECS, CS, CT
Ascending colon
5
Pathol Int
12
72
Male
CT, EGD
Stomach
6
Can J Gastroenterol
13
55
Male
CT
Ileum
ND
Surg Today
14
63
Female
US, CT
Ileum
2.5
Surg Today
15
73
Female
ECS, MRI
Sigmoid colon
ND
Arch Surg
16
63
Male
CT
Ileum
3
JSLS
17
85
Male
US, CT
Jejunum
4
J Gastroenterol Hepatol
18
62
Male
CT, CS
Sigmoid colon
3.5
Dig Dis Sci
19
55
Female
CT
Transverse colon
12
Am Surg
20
31
Female
CT
Ascending colon
5
Can J Surg
21
47
Female
US, CT
Ileum
5
Ulus Travma Acil Cerrahi Derg
22
56
Female
US, CS, CT
Transverse colon
5
Ulus Travma Acil Cerrahi Derg
23
64
Male
CS, CT
Transverse colon
6
Clin Gastroenterol Hepatol
24
55
Male
CT, ECS
Jejunum
4
World J Gastroenterol
25
42
Male
US, CT
Ileum
3
Case Rep Gastroenterol
26
47
Female
CT
Ileum
3
J Laparoendosc Adv Surg Tech
27
47
Female
CT, CS, Enema
Ascending colon
5
Endoscopy
28
36
Male
CS, CT, ECS
Ileum
9
Cases J
29
36
Male
CT, ECS
Ileum
4
J Nippon Med Sch
30
82
Male
CS, CT
Sigmoid colon
8
Gastrointest Endosc
31
69
Male
CT, CS
Transverse colon
7
Dig Dis Sci
32
38
Female
CS, CT
Ileum
3.3
Clin Gastroenterol Hepatol
33
38
Female
US, CT, CS
Cecum
6
Emerg Radiol
34
45
Male
CT
Ileum
2.5
N Engl J Med
35
43
Female
CS, CT
Ascending colon
5
Rev Esp Enferm Dig
36
57
Female
CS, CT
Transverse colon
5.5
Rev Esp Enferm Dig
37
51
Male
US, CT, CS
Ileum
3
Gastroenterology
38
77
Male
CT
Cecum
3.5
JSLS
39
46
Male
CS, CT, ECS
Descending colon
6
Endoscopy
40
33
Male
CT, CS, BE
Ileum
4
Case Rep Gastroenterol
41
32
Female
CT
Ascending colon
5.8
Gastroenterology
42
49
Male
US, CT
Descending colon
5
Gastroenterology
43
53
Female
US, CS, ECS
Ascending colon
7
Medicina (Kaunas)
44
26
Female
CT
Ileum
ND
Am J Surg
45
51
Female
CT
Transverse colon
6.2
J Gastroenterol Hepatol
46
68
Male
CS
Jejunum
3.2
World J Gastroenterol
47
52
Female
CT
Ileum
3.2
J Med Case Reports
48
62
Female
US
Ileum
7
J Clin Ultrasound
49
65
Male
CT
Ileum
1.2
World J Gastrointest Surg
50
68
Female
US, CT, ECS
Ileum
1.5
Surg Today
51 35 Male CT jejunum 6  


Article Categories:
  • Review

Keywords: Intussusceptions, Jéjunal lipoma, Intestinal tumor, Surgery.

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