Authors: Fatima Alhammadi, Amog Prakash, Fatma Mustafa Alhashimi, Maliha Jaffar, Faisel Ikram, Sara AlBastaki
Categories: Case Report, Case report, Ileo-ileal intussusception, Intestinal obstruction, Intussusception adult case, Intussusception in adults, Lipoma, Robotic surgery, Segmental resection, Small bowel obstruction
Source: International Journal of Surgery Case Reports
Intussusception in adults is rare, constituting a miniscule number of bowel obstruction pathologies. Clinical practice often considers it a last-resort diagnosis, as other causes of mechanical small bowel obstruction are more common. The diagnosis is a challenging one to make, as the “telescoping” motion of the intestines fluctuates in a waxing-and-waning nature. In adults, the etiology is predominantly a pathological lead point being either benign or malignant. When encountering an obscure cause of abdominal pain, intussusception caused by a small bowel lipoma may be the culprit.
In this unique case, we present the journey of a 55-year-old Emirati male who presented with severe abdominal pain, nausea and vomiting. The patient underwent various imaging modalities, initially an “incidental” lipoma finding in the terminal ileum until a diagnosis of ileo-ileal intussusception emerged in later imaging, given its unusual and fluctuating nature. Ultimately, a resection of the bowel segment with side-to-side anastomosis was done.
This case report aims to illuminate the diverse clues and incidental findings encountered during our patient's path to a diagnosis. By exploring the elements of this patient's journey to a diagnosis, we aspire to aid future clinicians in navigating the challenges of identifying obstructive pathologies and considering intussusception as a rare yet crucial differential diagnosis.
This case highlights the importance of a comprehensive approach to diagnosis and management of patients presenting with obstructive symptoms, incorporating both clinical insight and imaging modalities such as CT to ensure optimal patient outcomes. When confronting such an obscure cause of abdominal pain, intussusception caused by a small bowel lipoma may be the culprit.
Keywords: Intussusception in adults, Ileo-ileal intussusception, Intussusception adult case, Case report, Intestinal obstruction, Small bowel obstruction, Segmental resection, Robotic surgery, Lipoma
Adult intussusception accounts for 1% of all bowel obstructions [1]. Intussusception is a prevalent pediatric condition, unlike intussusception in adults, which ismuch less in incidence [3]. According to a retrospective review of adult intussusception, most pathological lead points are tumor-associated [4]. Intussusception occurs when one segment of the bowel “telescopes” into another segment of the bowel, resulting in a typical appearance of a “target” sign on the sagittal view of CT imaging [2,5]. Risk factors vary, the most significant risk factor being intestinal masses (benign or malignant). Other risk factors include post-surgical adhesions, endometriosis, fibroids, Meckel's diverticulum, inflammatory bowel disease, gastrostomy, or jejunostomy tubes [3,6]. Recognizing intussusception in a patient is highly challenging, as it usually presents with one of the most common complaints we encounter in intermittent abdominal pain [2]. Patients also present with other classic bowel obstruction nausea, vomiting, weight loss, and constipation [2].
Currently, there are no guidelines for optimal treatment of adult intussusception, but the consensus is surgical resection due to the high incidence of malignant lesions as pathological lead points [4]. This case report highlights the clinical presentation of a 55-year-old male experiencing obstructive symptoms attributed to ileo-ileal intussusception. The patient was managed in private hospital in Dubai, UAE. This work has been reported in line with the SCARE criteria [7].
A 55-year-old Emirati male, with no significant past medical history, presented to the emergency department with complaints of severe cramping abdominal pain in the periumbilical and right iliac regions for the past 4 h with a VAS score of 9/10. Associated with dry heaving, nausea and three episodes of non-bilious, non-bloody vomiting. The patient also noted that the pain has been present intermittently for the past three months but in a much milder form where it feels dull and predominantly right sided, which he attributed to indigestion. The patient denied any fever, dizziness, changes in bowel habits, bloody stool, difficulty passing flatus, unintentional weight loss, and urinary symptoms.
The patient was hemodynamically stable, visibly in pain but appeared in good health with no apparent signs of dehydration. Abdominal examination revealed a soft, non-tender, and non-distended abdomen. However, tenderness was elicited in the periumbilical and right iliac regions upon deep palpation. No masses or lumps appreciated upon palpation. When performing deep palpation, there was no lump or mass that was palpable. Furthermore, no guarding, organomegaly or ascites were appreciated upon examination.
Further exploring the patient's history revealed that a similar episode of these symptoms occurred less than a month ago in the UK. The patient was admitted to the emergency department and management involved IV morphine for symptomatic relief, the imaging done has rendered inconclusive results. Past surgical history of the patient is unremarkable. He has a BMI of 30.85 kg/m^2^.
Regarding the initial management, the patient was given IV fluids, ondansetron, PPI, and analgesia and placed on a liquid diet. Preliminary investigations ordered were a urinalysis and ultrasound of the abdomen and pelvis. Subsequently, the patient received a regimen consisting of paracetamol, tramadol, and morphine throughout the entirety of his hospital admission; however, these interventions failed to provide significant relief as the pain persisted. Our clinical assessment of the patient led us to consider several probable differential diagnoses, encompassing conditions such as acute gastritis, appendicitis, incarcerated or strangulated inguinal hernia, kidney stones, mesenteric ischemia, and various obstructive pathologies. Ultrasound of the abdomen and pelvis was normal. Urinalysis was mainly unremarkable except for positive ketones 1+ which can be attributed to dehydration. A complete blood count, urea and electrolytes, lipase and amylase, liver function tests, troponin I, D-dimer, and C-reactive protein were all within normal limits. Lactate levels were high at 2.9 mmol/L, which could point towards tissue necrosis due to bowel obstruction.
Subsequent to our review of the preliminary investigation findings, a CT KUB was ordered. It revealed a small left renal calcific density suggestive of a non-obstructing calculus, otherwise it was deemed normal (Fig. 1). An X-ray abdomen revealed small bowel distension and absence of ileocecal gas which is an indirect sign of bowel obstruction. CT angiography abdomen and pelvis with contrast revealed a small intramural lipoma in the terminal ileum which was thought to be an incidental finding (Fig. 2). No other obvious abnormality was seen to suggest small bowel ischemia or intussusception. The CT angiogram was rendered to be essentially negative.
Fig. 1 CT KUB small left renal calcific density suggestive of a non-obstructing calculus.
Fig. 2 CT angiography abdomen and pelvis with contrast revealed a small intramural lipoma in the terminal ileum.
Esophagogastroduodenoscopy and colonoscopy were performed on the third day of admission and yielded normal findings. However, it is important to note that the scope could not advance beyond the terminal ileum during the colonoscopy.
At this point after doing such exhaustive investigations, most imaging modalities showed no significant findings. Lastly, MR enterography was ordered. It revealed the characteristic donut/target sign indicative of intussusception (Fig. 3) found in the terminal ileum. The features seen were in keeping with ileo-ileal intussusception with a fatty lipomatous mass leading the intussusceptum. It is worthwhile to note that this mass is believed to be the same lipoma seen incidentally on the CT angiography scan done previously. The interloop adhesions seen also suggest a phenomenon of chronic intermittent intussusception with recent acute-on-chronic inflammatory response. This could explain the waxing and waning pattern of symptoms the patient has been experiencing.
Fig. 3 Donut/target sign typical finding of intussusception shown on the left with concentric rings appearance reflecting the layers of intussusceptum and intussuscipiens also shown on the right.
Given the established diagnosis and drawing insights from contemporary literature regarding the management of intussusception in adults, we decided to proceed with the resection of the bowel segment, encompassing the intussusceptum, intussuscipiens, and the pathological lead point. Consequently, four days after the initial encounter, the patient was scheduled for a robotic laparoscopic small bowel segmental resection with side-to-side anastomosis using the da Vinci robotic system. The primary operating surgeon is a consultant gastrointestinal surgeon with 20 years of experience in advanced laparoscopy and a fellowship in surgical gastroenterology. The surgical intervention unfolded as Upon robotic laparoscopic exploration, pneumoperitoneum was created with a Verres needle where 8 mm three ports were inserted. The pathology was visualized (Fig. 4) and can be described as ileo-ileal intussusception with a polypoid lesion causing invagination of the small bowel for almost 30–40 cm. The mass was intraluminal and submucosal lesion with a long stalk. The bowel was found to be easily reducible and viable. No mesenteric lymph node enlargement was found. Small bowel reduction then segmental resection was done by open laparotomy through a Pfannenstiel incision and Alexis wound retractor (Fig. 5). Resection was completed with a GI stapler and side-to-side stapled anastomosis was done (Fig. 5). The mesenteric defect was closed with PDS II 3/0 and wound closure was completed using stratafix. The excised specimen causing the invagination of bowel appeared as a lipomatous pedunculated mass with a long stalk (Fig. 6). The specimen was sent for histopathological diagnosing and was confirmed to be a small bowel lipoma.
Fig. 4 Ileo-ileal intussusception, pathological lead point lipoma shown by arrow.
Fig. 5 Left: ileo-ileal intussusception exposed by Alexis wound retractor. Right: side-to-side stapled anastomosis.
Fig. 6 Small bowel lipoma with long stalk as the pathological leading point for intussusception.
Following surgery, the patient experienced an uneventful and smooth recovery in the general surgical ward and oral fluids were resumed on day 1 postoperatively then gradually increased to soft diet on discharged on the third day post-operatively. Follow-up assessment was done 2 weeks later, revealing favorable progress and the patient reported being completely asymptomatic. The patient was informed of the histopathological findings' benign nature and provided reassurance regarding his overall health status.
Due to the intermittent clinical nature of the pathology, it can be a diagnostic dilema. We were privileged to have all the possible diagnostic modalities at our institution and with high index of suspicion, a diagnosis was established resulting in definitive treatment. It might remain undiagnosed outside a tertiary health facility and present as an acute surgical emergency with associated morbidity.
Among adults, intussusception is the least common cause of small bowel mechanical obstruction in the United States and Western Europe [2]. Data on the Middle East is scarce and yet to be properly established. The etiology behind intussusception varies between the pediatric and adult population, where in the latter it is mostly due to what is called a “pathological lead point” [3]. This pathological lead point can arise due to varying etiologies depending on the age of the patient. It could range from a lymphoid aggregate in the pediatric population to a benign or a malignant mass in adults. A case performed represents a recurrent fibrous polyp that predisposed a 46-year-old female to recurrent attacks of intussusception [8].
The pathophysiology of intussusception is rooted to the basic physiological peristalsis that occurs in our bowels. Any intestinal pathology that alters the usual peristaltic pattern predisposes the patient to intussusception. The continuity of the physiologic peristalsis further extends the invagination of the bowel [4]. The pathology has also been described as “telescoping” of the bowel into the other [5]. It can be explained to the patient as folding your socks or rolling up your sleeves. The terms intussusceptum and intussuscipiens have also been used in literature, where the intussuscipiens refers to the receiving end of the bowel and intussusceptum refers to the proximal part of the bowel which invaginates or telescopes into the distal end [9].
Classification of intussusception can be made based on either etiology (benign, malignant, idiopathic) or on location [10]. Classification of intussusception based on location is as (1) entero-enteric (2) colo-colic, (3) ileo-colic (4) ileo-cecal [10]. The most common site being the ileo-cecal junction [5].
Detecting intussusception based on clinical presentation is challenging and necessitates high clinical suspicion [3], as it presents with vague abdominal abdominal pain, nausea, vomiting, weight loss, constipation or diarrhea, hematochezia, and less commonly an abdominal mass [[2], [3], [4]].
Plain abdominal X-rays are usually done first line and may show typical findings of small bowel obstruction more distally [2] as we appreciated in our patient (absence of gas in terminal ileum). However, abdominal CT has always been regarded as the gold standard imaging modality for adult intussusception due to its high sensitivity as opposed to that of an MRI [11]. The findings seen on CT include the target or doughnut sign on sagittal view [2]. In our patient, we believe that the intussusception was transient, and the pathology was not present at the point of time when our initial CT KUB has been done. Due to the nature of the pathology, MRI enterography remains more sensitive in picking intraluminal small bowel pathology. CT scan will have a classic donut appearance during active intussusception but maybe missed when its reduced, however it was reported as an incidental lipoma in our case. The waxing and waning character of the pain should alert the clinician of this possibility. With regards to the use of diagnostic laparoscopy, it is an essential diagnostic tool at our institution in cases of unexplained abdominal pain and non-conclusive radiology. In this case it would have been the next diagnostic option if radiology was non-conclusive.
Most recommendations for management are rather straight forward and simple [3]. Early detection of the disease is fundamental to produce favorable outcomes and prevent complications that are sequalae to intestinal obstruction [3]. Initially, management in the emergency room is supportive. The mainstay of symptomatic treatment that we have similarly followed for our patient is a combination of analgesia, antiemetics, IV hydration, +/− nasogastric tube for decompression, and declaring NPO status if surgical intervention is likely [3].
In terms of treatment, it is the consensus that surgical treatment is the most definitive treatment when it comes to adult intussusception [6]. It is crucial to bear in mind that the more distal the mass is, the higher the probability of malignancy [12]. That is because etiologies of small bowel intussusception are mostly from benign lesions, whereas large bowel intussusception are more commonly found to be associated malignant lesions [12]. Due to the high stakes, and the possibility of tumor seeding through surgical manipulation [13], surgical resection is almost always preferred. Therefore, radiological findings of the lead point are what lead a clinician's chosen route of treatment [11]. While some argue that reduction is relatively safe when it comes to benign lesion, in an attempt to reduce the extent of resection and evade short bowel syndrome [6]. However, in the event of improper reduction, complications such as bowel perforation and failure of anastomosis may arise [12].
A retrospective study done on 88 adults with intussusception attempted to elucidate predictors of malignancy, and the following older age, colon-related intussusception, shorter intussusception length, higher maximum short axis diameter and more enlarged lymph nodes than in the benign group [14].
Chances of recurrence of intussusception are quite rare in cases of early intervention [11]. Possible complications of late diagnosis include bowel obstruction, bowel necrosis and as a result sepsis [3].
In conclusion, intussusception in adults, while infrequent, constitutes a crucial consideration for clinicians, particularly in cases where alternative etiologies remain elusive. It is also highly crucial to consider a history of benign incidental lesions as potentially problematic giving rise to intussusception in the context of abdominal pain. The utilization of baseline abdominal CT emerges as a valuable diagnostic tool, given its high sensitivity in ruling out and narrowing down probable causes of intestinal obstruction as compared to an MRI. Timely and accurate diagnosis is essential, as intussusception necessitates surgical intervention when the risks and benefits are carefully weighed. Recognizing its potential as a life-threatening emergency emphasizes the significance of swift medical attention and intervention. Nevertheless, it is worthwhile to keep in mind that, even though a malignant cause may be the most important cause of an intussusception in adults being diagnosed, there may be benign causes such as a lipoma to also consider.
Our on-site clinical research ethics committee has no ethical objection regarding this case report.
This work was supported and funded by the Emirates Society of Colon and Rectal Surgery for the publication of this case report. President of the society Dr. Sara Albastaki is an author of this case report, involved in conceptualization and reviewing the case report prior to submission.
Fatima Alhammadi: Writing – original draft. Amog Prakash: Data collection & Resources. Fatma Mustafa Alhashimi: Writing – Review & Editing. Maliha Jaffar: Project Administration and Editing. Faisel Ikram: Performed surgery and Supervision. Sara AlBastaki: Conceptualization and Funding acquisition. All authors reviewed and approved the final submitted manuscript.
Faisel Ikram.
Maliha Jaffar.
Fatima Alhammadi.
Not applicable.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.