05 December 2022: Articles
Adult Colo-Colonic Intussusception in the Setting of Invasive Mucinous Adenocarcinoma: A Case Report
Unusual clinical course, Unusual or unexpected effect of treatmentSayali Kulkarni1E*, Asama Rana2E, Karmina Choi3DEF
Am J Case Rep 2022; 23:e938124
BACKGROUND: Colonic intussusception is a very rare disease in adults, and if present, is usually manifested by another pathology, such as malignancy. This report describes the diagnosis and treatment of the underlying cause of intussusception, which was spontaneously reduced.
CASE REPORT: A 39-year-old woman with no significant past medical history presented to St. Joseph’s University Medical Center on July 2022 with gradually worsening abdominal pains for 1 year and hematochezia for 3 months. Physical examination was positive for left lower quadrant abdominal tenderness to palpation. A computed tomography scan of the abdomen and pelvis without contrast showed a long segment of intussusception involving the sigmoid colon and rectum, without any noticeable lesions. A repeat computed tomography scan with rectal contrast showed a 2.1×1.1-cm mesenteric mass in the sigmoid colon at the region of the intussusception. The patient was taken for a laparoscopic sigmoid resection with primary anastomosis, showing a 5-cm mass in the sigmoid colon, and surgical pathology confirming neoplastic etiology of intussusception. The patient recovered well after surgery, and was referred for oncological intervention soon afterward.
CONCLUSIONS: This report displays the importance of the type of imaging modalities with and without contrast to diagnosis and determine underlying causes of intussusception and further guide treatment options.
Keywords: Colectomy, Colonic Neoplasms, Intussusception, Sigmoid Neoplasms
Colonic intussusception is a very rare occurrence in adults and comprises 5% of all intussusception cases, with the remainder being in children; the overall incidence in adults is 1 to 3 cases per million per year [1,2]. Intussusception in adults is most commonly due to benign or malignant lesions, polyps, and strictures, which are mostly diagnosed in the intraoperative setting, with malignancy being the most common of the aforementioned conditions [3,4]. The mechanism behind intussusception is defined as a region of the proximal bowel telescoping within the distal portion of bowel, and rarely, the distal segment of bowel telescoping into the proximal segment . The definitive management of intussusception in adults involves treatment of the underlying cause of intussusception with surgical resection of the bowel involved . In this case report, we present a 39-year-old woman with a year-long history of abdominal pain, with associated hematochezia, and nausea. Colonic intussusception at the region of the sigmoid colon was diagnosed, with a confirmed underlying etiology of adenocarcinoma.
A 39-year-old woman with no significant past medical history presented to the Emergency Department for evaluation of acute worsening of her intermittent left lower quadrant abdominal pain. She stated the abdominal pain had been present for the past year, and in the last 10 days had been progressively worsening. The abdominal pain was associated with fatigue, loose bloody stools with mucus, and nausea. She denied any family history of colon cancer, Crohn disease, or inflammatory bowel disease. The physical examination findings were significant for left lower quadrant and suprapubic tenderness. The patient had a complete blood count, which showed anemia, and a basic metabolic panel and carcinoembryonic antigen (CEA) test, which were unremarkable. Owing to unavailability of contrast, a computed tomography (CT) scan of the abdomen and pelvis without contrast (Figures 1, 2) was performed, which showed a long segment of intussusception at the level of the sigmoid colon, with a characteristic target sign near the rectosigmoid junction. The surgical team was consulted. A repeat CT scan of the abdomen and pelvis with intravenous, oral, and rectal contrast (Figure 3) showed findings concerning for a 2.1×1.1-cm intra-luminal mass, as well as mass-like thickening of the wall of the sigmoid colon, thought to possibly be the lead point of the intussusception. Imaging also revealed a completely reduced intussusception, likely due to the rectal contrast administered. After her CT scan, the patient reported resolution of her abdominal pain. She was afebrile, hemodynamically stable, and in no acute distress. The patient was shortly discharged after careful observation, with plan for an outpatient colonoscopy and elective colectomy.
Unfortunately, before the patient could complete her follow up colonoscopy, she returned to the Emergency Department for recurring abdominal pain and bowel obstruction, similar to her initial presentation and likely due to recurrent intussusception. The physical examination was remarkable for diffuse lower abdominal tenderness. An abdominal X-ray series showed gaseous distension of the colon up to the site of the known sigmoid intussusception, concerning for partial obstruction or recurrent intussusception. Given the nature of her pain, previous imaging, concerns for malignancy, and large bowel obstruction, the patient was admitted for surgical intervention. She was scheduled for a laparoscopic sigmoidectomy. A preoperative colonos-copy was not performed due to inadequate bowel preparation.
During the surgery, the sigmoid colon was noted to be very redundant, with long mesentery, and the intussusception had reduced spontaneously. A large, firm 5-cm tumor was noted in the proximal-mid sigmoid colon. The tumor extended through the serosal surface, without evidence of perforation or invasion of adjacent structures. No mesenteric mass was visualized. The uterus, ovaries, and adnexa appeared normal, and no metastasis were noted upon visualization of the peritoneum and liver. Upon mobilization of the sigmoid colon and upper rectum from the lateral abdominal wall, there was significant mesenteric thickening and fibrosis, suggestive of chronic intussusception (Figure 4). A formal oncologic resection of the sigmoid colon was done with proximal lymphadenectomy and preservation of the left colic artery. The proximal transection point was at the level of the descending colon and sigmoid junction, with 8 cm of gross negative margin, and the distal transection point was at the border of the upper rectum. An end-to-end anastomosis was made with the descending colon and upper rectum. Surgical pathology of the resected sigmoid colon showed invasive mucinous adenocarcinoma with focal abundant signet-ring cells, with 5 of 21 lymph nodes positive for abundant mucin and occasional metastatic cells. One of the lymph nodes showed focal predominance of metastatic ring cells. The proximal inferior mesenteric artery lymph nodes taken at the root of the inferior mesenteric artery were negative for malignancy. Tumor, node, metastasis (TNM) staging was shown to be T3,N2a,M0, LVI+. Immunohistochemistry was indefinite for loss of PMH-2 expression, and no loss of nuclear expression of MLH-1, MSH-2, and MSH-6.
Her postoperative course was uncomplicated, and she made a good recovery and was discharged on postoperative day 2. She underwent oncological evaluation shortly after for further staging and adjuvant chemotherapy discussion.
Intussusception can be classified into primary or secondary intussusception. Primary intussusception occurs without an underlying lead point and mostly occurs within the small bowel . Secondary intussusception is caused by intrinsic masses or inflammatory conditions  and most commonly occurs within the small bowel. Although rare when compared with small bowel intussusception, colonic intussusception is mostly caused by malignant lesions . Clinically, symptoms of intussusception are nonspecific, and the mainstay of diagnosis for colonic intussusception is through CT imaging. CT remains the best imaging modality for recognition of the lead point and localization of any underlying masses . Treatment of adult intussusception itself has some controversy on whether to reduce the intussusception prior to resection; however, this carries a risk of seeding malignant cells to nearby structures, if malignancy is suspected. However, in the case of an idiopathic large bowel intussusception in an adult, there has been literature stating treatment with laparoscopic reduction is feasible, with an uneventful postoperative course . If the location of the intussusception is within the small bowel, there is an advantage to reduction, as it could prevent extensive resection of small bowel leading to short bowel syndrome . However, definitive treatment for adult intussusception is primary surgical resection. Right-sided intussusception typically requires resection and primary anastomosis. With left-sided intussusception to the rectosigmoid junction, the decision to do a re-section with primary anastomosis, diversion, or end colostomy depends on the intraoperative clinical status and degree of obstruction . Regardless, if the suspicion for malignancy is high, an oncological resection should always be performed.
This case highlights a unique presentation of colonic intussusception. The initial symptoms began as mild abdominal pain for a year, which acutely worsened, accompanied by 3 months of hematochezia. After initial CT scans confirmed colonic intussusception, a repeat CT with intravenous, oral, and rectal contrast showed a completely reduced intussusception with likely sigmoid mass. Due to symptoms consistent with large bowel obstruction, likely at the region of the intussusception, the patient was taken for operative intervention. Intraoperatively, there was no intussusception appreciated at the localized region the CT imaging demonstrated, giving this case its unique presentation of contrast-mediated reduced intussusception, followed by large bowel obstruction, due to underlying malignancy. Cases with left-sided, or rectosigmoid intussusception, can be treated with resection and colostomy, with Hartmann’s pouch, and anastomosis at a later time , but the present case was successfully treated with resection and primary anastomosis. Another unique factor in this case was the presentation of high-grade malignancy in a young patient with no risk factors for the development of colon cancer, no underlying family history, and negative tumor markers. However, this emphasizes that, based on clinical presentation, degree of obstruction, and imaging, the diagnosis of colon carcinoma should always be suspected regardless of risk factors.
Determining when to intervene with operative intervention and which surgical approach to consider should be guided by the case presentation, medical expertise, and current guidelines.
Intussusception in the adult population is a rarity, and prompt management should be conducted immediately to determine the underlying etiology of the intussusception. Colon carcinoma should always be suspected regardless of the patient’s lack of risk factors. Appropriate surgical management can be uniquely tailored to the patient, followed by adjuvant care in the setting of malignancy.
FiguresFigure 1.. Computed tomography of the abdomen and pelvis without contrast showing an arrow pointing to the region of colo-colonic intussusception. Figure 2.. Computed tomography of the abdomen and pelvis without contrast showing an arrow pointing to the region of colo-colonic intussusception. Figure 3.. Computed tomography of the abdomen and pelvis with intravenous, oral, and rectal contrast showing reduction of intussusception and an arrow pointing to the intra-luminal mass within the sigmoid colon. Figure 4.. Intraoperative photograph showing the mid-sigmoid colon, with arrows pointing at the borders of the mass, difficult to visualize otherwise.
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