A rare case of appendicular intussusception in an adult due to adenocarcinoma of appendix

Chitrawati Bal Gargade; Archana Hemant Deshpande

doi:10.4103/ijcs.ijcs_42_20

CASE REPORT

Year : 2021 | Volume : 4 | Issue : 3 | Page : 76-78

A rare case of appendicular intussusception in an adult due to adenocarcinoma of appendix

Chitrawati Bal Gargade, Archana Hemant Deshpande
Department of Pathology, Andaman and Nicobar Islands Institute of Medical Sciences, Port Blair, Andaman and Nicobar Islands, India

Date of Submission	16-Dec-2020
Date of Acceptance	06-Jun-2021
Date of Web Publication	24-May-2022

Correspondence Address:
Chitrawati Bal Gargade
Department of Pathology, Andaman and Nicobar Islands Institute of Medical Sciences, Port Blair - 744 101, Andaman and Nicobar Islands
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijcs.ijcs_42_20

Abstract

Intussusception is primarily a pediatric disorder and is infrequent in adults. Intussusception of the appendix is a rare disorder with much surgical curiosity. Here we present a rare case of adenocarcinoma of appendix that presented as appendiculocaecal Intussusception. This case is presented to address the importance of understanding clinical presentation, radiological findings, treatment modalities and histopathological examination in evaluating a case of appendicular intussusception in adult.

Keywords: Adenocarcinoma of appendix, adult, appendicular intussusception

How to cite this article:
Gargade CB, Deshpande AH. A rare case of appendicular intussusception in an adult due to adenocarcinoma of appendix. Indian J Colo-Rectal Surg 2021;4:76-8

How to cite this URL:
Gargade CB, Deshpande AH. A rare case of appendicular intussusception in an adult due to adenocarcinoma of appendix. Indian J Colo-Rectal Surg [serial online] 2021 [cited 2022 Jun 29];4:76-8. Available from: https://www.ijcrsonweb.org/text.asp?2021/4/3/76/345919

Introduction

Appendicular intussusception is a rare disease with the incidence of 0.01%.^[1] Primary adenocarcinoma of the appendix is also rare and mostly arises in preexisting adenoma. Intussusception in the adult is seen less frequently. It is most often secondary to a benign or malignant organic lesion. We report a rarer case of appendicular intussusception in adult due to adenocarcinoma of the appendix.

Case Report

A 39-year-old female patient presented to the emergency department complaining of a history of abdominal pain in the right iliac region with nausea and vomiting. The patient was afebrile. Physical examination revealed tenderness in the right lower quadrant. Laboratory studies were all within the normal limits. Contrast computed tomography (CT) showed ill-defined soft-tissue mass adjacent to the terminal ileum with suspicion of ileocaecal intussusception. Intraoperative findings revealed ileoileal and ileocaecal intussusception and mass in cecum. Appendix could not be found. Right hemicolectomy was done with ileocolic anastomosis.

The resected specimen showed ileoileal and ileocaecal intussusception with complete inversion of appendix into cecum. The inverted appendix measured 5 cm in length [Figure 1]. A proliferative growth was seen involving the base and wall of appendix [Figure 2]. Microscopic examination showed tubulovillous adenoma [Figure 3] with foci of well-differentiated invasive adenocarcinoma [Figure 4].

Figure 1: Resected colectomy specimen showing inverted appendix

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Figure 2: Sessile growth involving appendix

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Figure 3: Microscopy (H and E, ×40) showing tubulovillous adenoma with normal epithelium

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Figure 4: Invasion of muscularis propria by adenocarcinoma (H and E, ×100)

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Discussion

Intussusception is defined as the invagination of a bowel loop with its mesenteric fold (intussusceptum) into the lumen of a contiguous portion of the bowel (intussuscipiens). This is often due to lesions within the bowel lumen acting as “lead points” during normal peristalsis. Intestinal intussusception in adults is uncommon, accounting for 5% of all intussusceptions and representing only 1% of intestinal obstructions.^[2]

Small bowel intussusception is more common still appendicular intussusception is very rare. Since the first report of intussusception of appendix by McKidd in 1858, very few cases have been reported.^[3]

According to some case series, appendicular intussusception is predominantly a pediatric condition, with most cases observed in the first decade of life, with the average age of occurrence being 16-year-old.^[4] While other studies showed that appendicular intussusception is more commonly encountered in the age group of adults, with a frequency which is >2 times higher than that of the pediatric age group. Interestingly, intussusception of the vermiform appendix happens more often in the male population in the pediatric age group, while female patients are more in the age group of adults.^[5] Our patient was a 39-year-old female The presentation of adult intussusception varies from asymptomatic to chronic pain. The patients suffering from appendicular intussusception may have variable symptoms and signs ranging from acute appendicitis to chronic abdominal pain, palpable abdominal mass, rectal bleeding, and constipation. Therefore most of the time patient receives either symptomatic treatment and diagnosis may get delayed and the patient presents with intestinal obstruction when the disease is advanced. Our patient presented with acute abdominal pain, fever, and vomiting.

Ultrasonography plays an important role as a diagnostic method for appendicular intussusceptions. Reported radiologic signs of intussusception include:

Ultrasound-multiple concentric ring sign/target-like appearance
Barium enema-coiled-spring sign and caecal filling defect with nonfilling of the appendix
CT-well-demarcated cylindrical mass of soft tissue
Colonoscopy-mushroom like polypoid tumor with the dimple on top

Intussusception in adults differs from those in children in various aspects. Most of the appendicular intussusceptions seen in children are due to anatomical variations. In adults, the majority of cases have a definite underlying cause, with primary intussusceptions accounting for only 15%–25%.^[6] Secondary causes of intussusception in adults include foreign bodies, fecoliths, parasites, cystic fibrosis, lymphoid hyperplasia, endometriosis, mucocele, polyps, adenocarcinomas.^[7] Children with intussusception present with the classic triad of vomiting, rectal bleeding, and abdominal pain while the change in bowel habits, constipation, or abdominal distension are the nonspecific symptoms and signs of intussusception in adults. The treatment in children is mostly nonoperative (barium hydrostatic reduction) while in adults surgical resection is almost always required.

Adenomas of the appendix account for approximately 0.02%–0.14% of appendiceal lesions.^[8] Although most appendicular adenomas are detected incidentally in appendectomy specimens, cases associated with intussusception have also been described in case reports.^[7],[9]

Appendiceal villous adenomas pose a risk of progression to invasive carcinomas such as other villous adenomas of the large bowel. Primary adenocarcinoma is a very rare entity, which in most cases arises from a preexisting adenoma.^[7] Only few cases of rare combination of appendiceal intussusception induced by primary appendiceal adenocarcinoma in adenoma similar to our case are reported till date.^{[9],[10],[11]}

Surgical treatment varies from appendectomy to right hemicolectomy depending on the etiology and the degree of intussusception. Appendicular intussusception especially in younger patients may have a benign, physiological cause, and laparoscopy with reduction may be an acceptable strategy. Ninety percent of appendicular intussusception in adults harbors a pathological process. Surgical management remains the mainstay treatment modality for a majority of adult patients with appendicular intussusception. In the present case, intussusception right hemicolectomy was done as intussusception was nonreducible and also showed a mass in cecum.

To the best of our knowledge, this is the only first case of adult appendicular intussusception secondary to adenocarcinoma of appendix in villous adenoma reported till date in Indian literature.

Conclusion

In adult intussusception chronic abdominal symptoms are more common than acute symptoms. AI should be considered in the workup of chronic abdominal pain in women
Adenocarcinoma of appendix can present as appendicular intussusception and treatment is different from intussusception due to nonmalignant lesions. Hence, complete radiological evaluation should be done before deciding surgical strategy.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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