
==== Front
Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)01023-X
10.1016/j.ijscr.2024.110242
110242
Case Report
Pedunculated colonic lipoma causing adult colo-colic intussusception: A case report and literature review
Bacha Dhouha a
Kammoun Neirouz neirouzkammoun@gmail.com
b⁎
Mallek Ines a
Gharbi Lassad b
Lahmar Ahlem a
Slama Sana Ben a
a Pathology department, University Hospital Mongi Slim, La Marsa, Tunisia
b Surgery department, Mongi Slim Hospital, Tunisia
⁎ Corresponding author. neirouzkammoun@gmail.com
07 9 2024
10 2024
07 9 2024
123 1102427 8 2024
29 8 2024
4 9 2024
© 2024 Published by Elsevier Ltd on behalf of IJS Publishing Group Limited.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction

Colo-colonic intussusception (CI) in adults is rare, usually caused by malignant conditions. Nonmalignant tumors, like colonic lipomas (CLs), can also be an underlying cause.

Case report

We report an unusual case of a 62-year-old man admitted to the emergency department with acute abdominal symptoms.

The CT scan confirmed the colonic obstruction, causing significant distention in the transverse and right colon. It also revealed an intraluminal pedunculated colonic mass with fatty density. Peroperatively, a descending colon intussusception was noted. We performed a left colon resection with a double colostomy on the left flank. The postoperative follow-up was uneventful.

Pathologic examination of the surgical specimen revealed two lipomas. One of them was pedunculated and protruded into the colonic lumen causing the intussusception.

Discussion

We conducted a literature review of adult CLs complicated by CI, covering the period from January 1900 to June 2024, including 203 cases. We excluded lipomas exclusive to the small intestine and ileocecal valvula. Our analysis focused on the clinical and pathological characteristics of these cases, as well as the available management options.

Conclusion

Colonic intussusception due to lipomas are uncommon with a challenging preoperative diagnosis despite the evolution of imaging procedures. We aimed by our case to highlight such pathology and to study its features and the possibilities of its management.

Highlights

• Acute colonic intussusception in adults due to lipoma is uncommon.

• Symptoms are nonspecific, often presenting as abdominal pain, subocclusive syndromes or even acute obstruction.

• Preoperative diagnosis of CI due to CLs is difficult, despite the evolution of imaging procedures.

• Lipoma's size and clinical presentations determine the therapeutic approach.

Keywords

Colonic lipoma
Intussusception
Surgery
Endoscopic resection
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pmcThe case report is reported in line with the SCARE criteria [1].

1 Introduction

Intestinal intussusception is rare in adults and the overall incidence is around 2–3 cases per 1,000,000 of the general population annually, or 1.86 cases/year [2]. It is associated with lead points affecting the colon in around 17 % of patients [2].

A lead point of colo-colonic intussusception (CI) is found in 70–90 % of adult intussusceptions, contrary to pediatric conditions, which is idiopathic in 90 % of cases. The lead point is malignant in 30 to 65 % of the cases [2,3]. Pathological conditions like carcinoma, lymphoma, diverticulum, and adenomatous polyp act as a lead point and produce invagination within the lumen, thus predisposing to intussusception.

In the other hand, colonic lipomas (CLs) are uncommon in adults. They are the second most common benign tumor in the colon after adenomatous polyps, with an incidence of only 0.2 % to 4.4 % of patients undergoing endoscopy [4]. often asymptomatic, however, around 25 % of patients may develop symptoms. When a giant lipoma is present, patients who have symptoms reach 75 % of cases. These symptoms include bleeding, pain, or mimic malignancy [5].

They could even lead to life-threatening complications such as perforation or intussusception (CI), where one segment of the colon telescopes into another [6,7].

We aim by our work to present an unusual case of multiple CLs in an adult, that resulted in descending colon invagination and acute abdominal symptoms.

Informed consent was obtained from the patient for the publication of this report.

We also realized a literature review of cases of CLs complicated by CI. We report the different clinical and pathological presentations of these tumors, along with the various management options that are considered.

2 Material and methods

We report a case of a 62-year-old man with no medical history as an example of an unusual colonic intussusception caused by a lipoma. Then, we realized a literature review of all similar cases from 1900 to June 2024 regardless of language. Our research used the Medline database.

A total of 180 articles were included, out of 316 records; 208 patients were involved (Fig. 1). Different clinical and pathological presentations of these tumors, along with the various management options that are considered are reported by our work.Fig. 1 Flowchart for article inclusion.

Fig. 1

3 Case report

A 62-year-old man with no medical history, presented to the emergency department with abdominal pain, distention, and vomiting. On the physical examination, there was slight abdominal distension with tenderness in the left hypochondrium. There was no palpable mass. A digital rectal exam revealed an empty and collapsed rectum. Routine lab tests showed normal results. A contrast-enhanced CT scan was performed, revealing a left colon obstruction, due to an intraluminal colonic mass with fatty density. The mass measured 5 × 3.5 cm ∗ 6.5 cm. Consequently, the obstruction has led to fecal stasis in the transverse and right colonic areas (Fig. 2).Fig. 2 Contrast-enhanced CT scan: intraluminal colonic mass with fatty density measuring 6.5 cm in its long axis (arrows) with colonic distension. Axial (A) and coronal (B) images.

Fig. 2

Initially, a laparoscopic approach was attempted, revealing CI at the level of the descending colon (Fig. 3). Dissection and liberation of the left colon were challenging due to the great colonic distension, requiring conversion by a midline laparotomy. We performed a segmental resection of the left colon taking off the intussusception with a double colostomy on the left flank, as the Bouilly-Volkmann procedure. Pathologic examination of the surgical specimen showed a prolapsing pedunculated, soft, yellow, and well-defined tumor that measured 5x4cm. This tumor was causing the CI (Fig. 4). Additionally, a second 2x1cm fatty formation was found 2 cm away from the primary tumor in the subserosa.Fig. 3 Per operative imaging: Colo-colic intussusception of the descending colon which is distented. The collar is narrow (arrow) (A) and the tumor protruded into the colon lumen. (arrow) (B).

Fig. 3

Fig. 4 Macroscopic features: A well limited and budding tumor with a yellow cut surface (A) Tumor surface is smooth and well defined (B). (For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.)

Fig. 4

Histological analysis confirmed that the two masses were benign colonic neoplasms composed of mature adipose tissue. The adipocytes were arranged in lobules separated by thin fibrovascular septa. The adipocytes were regular and uniform in size, without any cytonuclear atypia (Fig. 5, Fig. 6).Fig. 5 Histologic features: Submucosal proliferation of mature adipocytes dissociated by thin fibrous septa. The mucosa is atrophic and hemorrhagic (arrows) (Hematoxyllin Eosin x 40) (A) Adipocytes are regular and uniform in size (arrows) (Hematoxyllin Eosin x 200)(B).

Fig. 5

Fig. 6 The second lipoma: a sub serous 2 cm yellow lipoma (A) well limited (lines) (B) and composed of sheets of mature adipocytes (arrows) (Hematoxyllin Eosin x 40)(C). (For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.)

Fig. 6

No atypical cells or myxoid changes were noted within the fibrous septa. In the submucosal lipoma, the mucosa was atrophic, hemorrhagic, and focally ulcerated (Fig. 5). Postoperatively, the patient had an uneventful recovery and was discharged after four days. Continuity was restored after 3 months. There was no evidence of recurrence at the one-year follow-up.

4 Discussion

Our literature review included cases of adult CLs complicated by CI. A total of 180 full-text articles including 208 patients were analyzed. The articles consisted mainly of retrospective case reports, and some case series [[8], [9], [10], [11], [12], [13], [14], [15], [16], [17], [18], [19], [20], [21], [22]].

Demographic data were available for 203 patients. The median age of the patients was 51.57 (19–88) years. The distribution of patients by age groups is illustrated in the Fig. 7.Fig. 7 Distribution of patients by age groups.

Fig. 7

59 % of the patients ranged between 41 and 60 years. There were 88 males and 115 females with a female-to-male sex ratio of 1.3.

For symptoms, most of CLs are asymptomatic, but around 25 % of patients developed symptoms [9]. On the other hand, when a giant lipoma is present, patients who have symptoms reach 75 % of cases [9].

In our review, abdominal pain was the most common symptom (83.17 %) sometimes accompanied by abdominal distension (14.85 %) [[8], [9], [10],[21], [22], [23], [24], [25], [26], [27], [28], [29], [30]]. The other symptoms are detailed in Table 2.

Physical examination usually revealed abdominal tenderness, which may vary depending on the affected colonic segment by the intussusception, occurring in the upper or lower quadrants [2,9,10,32].

In rare cases, an abdominal palpable mass was noted [7,25,31,[33], [34], [35]]. This mass prolapsed or was palpable in the digital rectal exam in only 4 cases [5,29,36,37].

For the location, CLs are usually located in the transverse colon in 28 %, followed by the sigmoid in 20 %, the caecum in 18 %, and the rectum in 4 % of the cases in a systematic review published by Tasseli et al. [38], (Table 1).Table 1 Location of colonic lipomas complicated by intussusception in the literature review (1900–2024).

Table 1Locations	N/208	%	
Transverse colon	58	27.89	
Sigmoid	40	19.24	
Cecum	37	17.78	
Ascending colon	39	18.75	
Descending colon	27	12.98	
Rectum	7	3.36	

Table 2 Clinical symptoms of colonic lipomas complicated by intussusception in the literature review (1900–2024) in 202 patients.

Table 2Symptoms	N/202	%	
Abdominal pain	168	83,17	
Constipation	43	21.28	
Vomiting	33	16.33	
Rectal blood loss	32	15.84	
Distension	30	14.85	
Diarrhea	20	9.9	
Hematochezia	21	10.39	
Weight loss	24	11.88	
Nausea	21	10.39	
Obstruction	20	9.9	
Mass prolapsing through the anal canal	15	7.42	
Altered bowel movements	13	6.43	
Rectal mucus and blood loss	8	3.96	
Tenesmus	5	2.47	
Bloating	6	2.97	
Fever	6	2.97	
Alterated diarrhea/constipation	6	2.97	
Dyspepsia	1	0.49	
Chest tightness and Breathing difficulties	1	0.49	
Fatigue	1	0.49	
Anorexia	1	0.49	

Table 3 Treatment of colonic lipomas complicated by intussusception in the literature review (1900–2024) in 208 patients.

Table 3Treatment	N/208	%	
Endoscopic polypectomy	23	11.05	
Open polypectomy	14	6.73	
Right colectomy	67	32.21	
Extended right colectomy	9	4.33	
Segmental colectomy	41	19.71	
Left colectomy	26	12.5	
Sigmoidectomy	19	9.14	
Hartmann sigmoidectomy	3	1.44	
Anterior rectal resection	6	2.89	

Lipomas are the third most common benign colon tumors, after hyperplastic and adenomatous polyps [39]. While solitary lipomas were the most common, they were multiple in about 10 % of the cases, as in our case [25].

In this context, it is uncertain whether the proximity of multiple CLs increases the risk of intussusception through mechanical mechanisms.

In a single lipoma whose size and weight increase through time, the distortion of the colonic wall or the luminal obstruction can create a pressure gradient and alters normal peristaltic activity, pushing the affected segment of the colon into the adjacent one, causing an invagination [40].

All the added circumstances leading to an increase in abdominal pressure, like chronic constipation, and congenital or acquired dolichocolon, which corresponds to unusually lengthened bowel, forming extra loops, kinks and tortuosities, may be considered as risk factors for this complication [41].

Their slow growth often leads to a delayed onset of symptoms [4]. Therefore, they are usually asymptomatic and only discovered incidentally during colonoscopies, surgeries, or autopsies [5].

Symptoms include non-specific abdominal pain, lower intestinal bleeding, changes in bowel habits, nausea, constipation, diarrhea, anemia, reduced appetite, weight loss, and even life-threatening complications such as colon stenosis, obstruction or intussusception [5]. CI was found in 17 % of CL cases [7].

Unlike the intussusception of the small intestine in which benign lesions are responsible for most cases, CI in adults is secondary to malignant etiology in 65 % of the cases. Therefore, it is appropriate to rule out a malignant pathology [7].

Pre-operative diagnosis may be difficult [42]. The instrumental methods used to identify CI underlying CLs were ultrasound, abdominal X-ray, barium enema, CT scan, and colonoscopy. They should be done urgently, with rapid surgical intervention as a priority to prevent extensive colonic necrosis. Plain abdominal X-rays may show a bowel radiolucent area indicating the presence of a lipoma if it contains enough fat [43,44].

Barium enema studies are often not helpful for making a diagnosis, but they may show a changing mass in size and shape during the exam, known as the “squeeze sign” [45].

Ultrasonography can be helpful, especially if there is a noticeable mass.

An abdominal CT scan is the preferred noninvasive radiologic modality for diagnosing intussusception from CLs [46].

It is highly effective in detecting CI; however, it may be less sensitive in identifying the exact origin of the condition. Its sensitivity to diagnose intussusceptions has been reported from 71.4 % to 87.5 % while its specificity in adults has been reported to be 100 % as verified by the subsequent surgery [47].

Intestinal intussusception presents two classic images: the “sandwich” image in the longitudinal section, depicting the head of the intussusception, and the “cocarde” image in the transverse section, showing the intussusception mass [7].

The fat density and smooth borders can identify CL. A “target sign” on sagittal views and a “sausage-shaped mass” on axial views can be indicative [7]. “Target sign” refers to a characteristic appearance, where the structure resembles a target with alternating rings of different densities. “Sausage-shaped mass” describes the appearance of the affected intestine, where it appears elongated and bulging, resembling the shape of a sausage.

CT scan can appear heterogeneous, reflecting the degree of infarction and fat necrosis present at the time of radiologic evaluation [48].

In our case, it was an intraluminal colonic mass with a fatty density that caused fecal stasis in the transverse and right colonic areas (Fig. 2).

Abdominal magnetic resonance imaging is an alternative in the diagnosis and shows a signal intensity characteristic of adipose tissue on T1-weighted sequences [49].

It can indicate the presence of intussusception and identify the lipoma, which can be distinguished from cancer and other types of growths [32].

The colonoscopy is only able to detect submucosal CL by directly visualizing the mass, which appears as a yellow and elastic mass [50]. The elasticity of the mass can be confirmed by observing the “cushion or pillow sign”, characterized by the tumor easily compressed and released when some closed biopsy forceps is used [25]. Another characteristic sign is the “naked fat sign” which involves the extrusion of fat after a biopsy of the colonic mucosa [51].

Regarding treatment, authors recommend surgical intervention as the primary approach for complicated lipomas, especially for sessile lipomas, where endoscopic removal would be technically challenging [52].

They also recommend surgery for lipomas greater than 2 cm, even without complications, especially in older patients is whom malignancy is always suspected [52].

Hydrostatic barium enemas have been used as a primary approach for CI. Successful reduction typically ranges from 50 % to 75 %, with recurrence rates falling between 8 % and 20 % However, there is a risk of perforation [48].

Colonoscopic resections are usually avoided during an acute presentation due to the inability to prepare the bowel and the uncertainty about the possibility of colonic necrosis, which may increase the risk of perforation [9]. Attempts to reduce intussusception through colonoscopy have been very rarely successful [53]. However, some authors have reported that pedunculated and sessile lipomas can be removed endoscopically without causing perforation [9]. For this purpose, various endoscopic techniques have been used, such as endoloop ligation, mucosal and submucosal resection, and deroofing of the lipoma [9].

Okada et al. used curative endoscopic treatment for CI caused by a giant CL, employing a wedged balloon and ligation with detachable snares [54]. An effective hybrid technique was performed associating endoscopic unroofing and mucosal resection for a large colonic lipoma with intussusception [55]. For distal colorectal lipomas, transanal resection may be a safe and efficient treatment method, with the intussusception resolving spontaneously [37].

Surgery resection depends on suspicion of malignancy, with priority given to en-bloc removal.

When a lipoma diagnosis is made pre-operatively, the surgery depends on the size of the tumor, its location, the length of intussusception and the extent of potential necrosis [25].

Then, the primary reason for surgical treatment is to prevent the possibility of overlooking a malignant tumor, particularly in elderly patients and in the presence of necrotic mucosa, ulceration, and bleeding [9,24]. In addition, reports of endoscopic treatment for CL complicated by CI are mostly from 2022 and 2023 [9,37,53].

Therefore, the experience with these techniques is relatively recent and uncommon worldwide, with selective indications at times. For instance, the application of endoscopic procedures is confined to tumors originating from the submucosa, as the risk of perforation increases in deeply located lipomas [9]. Furthermore, the location of the lesion also plays a significant role, as right-sided colon lesions are more susceptible to perforation than left-sided colon lesions [9].

In the literature review, right colectomy and segmental colectomy were the two common treatments, with respectively 32.9 % and 19.9 % of the cases. The other treatments are detailed in Table 3.

The definitive diagnosis is obtained through a pathology examination, which confirms the absence of malignancy. Some authors recommend an intraoperative frozen section to avoid unnecessary radical resection and improve patient prognosis [56].

When CLs became complicated, their average size was 5.9 cm in length, 4.5 cm in width, and 3.4 cm in height with a minimum of 15 × 15 × 15 mm3, and a maximum of 160 × 110 × 100 mm3. The largest lipoma measured 16 × 11 × 11 cm and it was located at the level of the ascending colon, necessitating a subtotal colectomy [57]. The distribution of tumor size in relation to 4 cm, based on available data, is depicted in the Fig. 8.Fig. 8 Distribution of tumor size relative to 4 cm according to available data.

Fig. 8

Histologically, about 90 % of CLs develop from the submucosa layer of the colon, and the remaining 10 % originates from subserosa, particularly from an appendix epiploica [25]. Intramucosal lipomas are even rarer, with the possibility of an associated Cowden syndrome [58].

The colonic lipomas are sessile in 90 % of the cases [24]. They can rarely be pedunculated as in our case and several documented cases in the literature.

Typically, the lipoma is composed of mature adipocytes of uniform size without cytological atypia. The diagnosis can be challenging due to the presence of atypical features such as an expansive growth pattern, dissociating the muscularis propria and/or the serosa [8,59]. Moreover, lipomas can also present atypical stromal cells, cellular fibrosis, florid vascular proliferation, and increased mitotic activity due to repeated intussusception, mimicking a primary angiomatous lesion or malignancy [8,9,15,60].

The mucosa overlying the lipoma may be eroded, ulcerated, ischemic, regenerative, necrotic or atrophic, and hemorrhagic as in our case. This is due to chronic pressure and violent peristalsis effects caused by intussusception. In some cases, it can be polypoid [5,60].

Concerning differential diagnosis, it's crucial to consider conditions that mimic colonic lipomas.

If the CT scan shows prominent fibrous septa and nodularity are evident, the most important potential alternative diagnosis is a well differentiated liposarcoma [61].

A fluorescence in situ hybridization (FISH) analysis for MDM2 gene was performed in one case to rule out this diagnosis [62].

This condition is rare comparing to more common benign colonic conditions such as adenomatous polyp, complicated diverticulosis, neurofibroma, hemangioma and leiomyoma [25]. Lipomatous hypertrophy of the ileocecal valve, for instance, shares a benign nature but lacks the well-circumscribed appearance typical of lipomas [63].

Another consideration is pseudolipomatosis, which is an artifact induced by air insufflation or disinfectant use during endoscopy. Microscopically, it presents with numerous small rounded empty spaces devoid of nuclei and typically tests negative for S100 protein [64].

Lipomas can also mimic a primary angiomatous lesion if a florid vascular proliferation is present.

[60].

In all cases, there were no recurrences of the lipoma. Postoperative complications are very rare and comparable to those reported after open procedure for intestinal obstruction due to benign condition. Patients were discharged after a period of 2 to 18 days [60]. Complications include intra-abdominal abscesses, postoperative ileus and wound infection [9]. Mortality from CI in adults increases from 8.7 % in benign causes to 52.4 % in malignant causes [65].

5 Conclusion

Our case describes a rare occurrence of two lipomas in the descending colon. One of them was large, pedunculated and causes CI. The second was in the subserosa and located 2 cm away from the first.

Preoperative diagnosis of CI due to CLs is difficult, despite the evolution of imaging procedures. Abdominal CT scan is the method of choice for the diagnosis.

Lipoma's size and clinical presentations determine the therapeutic approach. However, in most cases, colonic resection is the best therapeutic option. Endoscopic resection is increasingly performed using different methods, effectively and without complications.

This case and the literature review highlight the importance to consider lipoma among the causes of large intestinal intussusception in adults.

They standardize the necessary procedures required for the diagnosis and treatment of this kind of lesion.

Ethical approval

Our institutions “Mongi Slim Hospital” and “School of Medicine of Tunis” require no ethical approval for case reports. It is required Author Form for studies on human participants. This is just a case report with written patients approval.

Funding

None.

Author contribution

Kammoun Neirouz: conceptualization, data curation, redaction.

Bacha Dhouha: conceptualization, data curation, redaction.

Ines Mallek: conceptualization, redaction.

Lassad Gharbi: resources, visualization.

Ahlem Lahmar: resources, validation, visualization.

Sana Ben Slama: supervision, visualization.

Guarantor

Kammoun Neirouz.

Research registration number

Not applicable.

Conflict of interest statement

All authors declare that there is no conflict of interest.

Acknowledgment

None.

We had the consent of the patient to publish the work.
==== Refs
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