Coexistence of cecal duplication cyst and Meckel's diverticulum presenting as intestinal obstruction: A case report

Abstract

Introduction

Enteric duplication cysts (EDC) are rare anomalies of the gastrointestinal tract, with only 0.4 % occurring in the cecum. Meckel's diverticulum (MD) is a common congenital anomaly affecting up to 2 % of the population. The simultaneous occurrence of these two conditions is rare with no existing guideline on treatment.

Case presentation

An 11-month-old boy presented with fever, vomiting, and abdominal distension. A contrast-enhanced computed tomography scan confirmed the diagnosis of an enteric duplication cyst causing intestinal obstruction. The patient underwent exploratory laparotomy, during which a cecal duplication cyst measuring 30 × 20 mm was found along with MD in the distal ileum. Right limited hemicolectomy was performed. Histopathological examination revealed features consistent with an enteric duplication cyst and the presence of gastric mucosal heterotopia.

Clinical discussion

Differentiating EDC from MD is a significant challenge, as both can present with similar symptoms and be positive on a Tc-99 m radionuclide scan. The final diagnosis of EDC and MD can only be made by correlating the imaging findings with the surgical findings and pathological features.

Conclusion

Cecal duplication cysts should be considered a differential diagnosis in infants who present with intestinal obstruction. Although their presentation may resemble that of Meckel's diverticulum, both conditions can coexist. Excision of duplication cysts in children is considered a safe and efficient treatment approach.

Highlights

• •Enteric duplication cysts in the cecum are rare, accounting for only 0.4 % of cases.
• •Cecal duplication cysts should be considered in infants with intestinal obstruction.
• •Despite resembling Meckel's diverticula in presentation, both conditions can coexist.

1. Introduction

Enteric duplication cyst (EDC) is a rare congenital anomaly with an incidence of approximately 1 in 4500 livebirths [1,2]. It can affect any segment of the gastrointestinal tract (git), with the ileum being the most common site, followed by the mediastinum, colon, stomach, duodenum, rectum, esophagus and cervix [3]. Cecal duplication cysts (CDCs) are very rare, accounting for 0.4 % of all EDC [4].

Meckel's diverticulum (MD) is the most common congenital anomaly of git, occurring in up to 2 % of the population. Both EDC and MD are more common in boys than in girls and usually present in infancy or early childhood. The presentations of EDC and MD can mimic one another, as both can present with abdominal pain, lower gastrointestinal bleeding, intestinal obstruction and an abdominal mass. The diagnosis of EDC and MD is made with the help of ultrasound (USG), computed tomography (CT), and technetium 99 m radionuclide scans, which can help to identify the location, size, and contents of the EDC and MD. In some cases, a definitive diagnosis can be made only during surgery; hence, several previous studies have reported a misdiagnosis of EDC as MD [5,6].

The simultaneous presence of EDC and MD is very rare and has been reported in only few prior studies [7,8]. This paper, adhering to the SCARE guidelines, details a unique case of CDC and MD in an 11-month-old boy managed successfully with right limited hemicolectomy [9].

2. Case presentation

An 11-month-old male presented with fever and vomiting for four days, accompanied by abdominal distension for three days. The fever was continuous, with a maximum recorded temperature of 102 °F, and vomitus containing recently ingested food particles occurring 3–4 times/day. He developed abdominal distension with an increase in the frequency of defecation up to 4–5 times/day, with stools that were foul smelling and not mixed with blood. There was no history of trauma, recent illness, loss of consciousness, yellowish discoloration or constipation. He was delivered at term via vaginal delivery with a birth weight of 3.5 kg. The patient's immunization status was up to date. The patient was alert, and his vital signs were within normal limits except for temperature of 101 °F. Abdominal examination revealed a soft and distended abdomen without organomegaly. The remainder of the systemic examination findings were unremarkable.

Laboratory investigations revealed a reduced hemoglobin of 7.2 g% (normal 12–18), increased platelet count of 534,000/mm³ (normal 150,000–400,000). White cell count, renal function tests, liver function tests, coagulation assays, routine urine and microscopic examinations, and parasitology were normal. Abdominal X-ray revealed multiple air-fluid levels suggestive of intestinal obstruction (Fig. 1). Ultrasonography (USG) of the abdomen and pelvis revealed a 35 × 25 mm well-defined, anechoic structure with internal echoes in the right upper quadrant. Contrast-enhanced computed tomography (CECT) of the abdomen and pelvis revealed a well-defined intraperitoneal cystic lesion measuring approximately 30 × 22 × 26 mm with post contrast enhancement in the right upper quadrant of the abdominal cavity, closely related to an adjacent bowel loop without calcifications, septa, or solid components (Fig. 2). In addition, the prominence of the large bowel and small bowel loops with maximum diameters of 3 cm and 2 cm, respectively, was noted, indicating intestinal obstruction. Based on these findings, a diagnosis of a CDC causing intestinal obstruction was made.

Fig. 1.

Erect abdominal X-ray showing multiple air fluid levels suggestive of intestinal obstruction.

Fig. 2.

CECT scan of abdomen and pelvis showing cystic lesion in right upper quadrant of abdomen (white arrow).

The patient was kept nil per os, resuscitated with intravenous fluids and underwent exploratory laparotomy. An intraoperative cyst measuring 30 × 20 mm was identified at the mesenteric border of the cecum (Fig. 3), confirming a cecal duplication cyst. The small intestine was distended without necrotic changes. Right limited hemicolectomy with ileocolic anastomosis was performed. In addition, an MD of 1.5 cm with a wide base was identified at the distal ileum, which was not resected (Fig. 4). The postoperative course was uneventful, and the patient was discharged on the 3rd postoperative day. Histopathological examination confirmed an enteric duplication cyst, without communication to the intestinal lumen. Within the cyst wall, both the intestinal mucosa and muscularis propria were observed, along with the ectopic gastric mucosa. He was asymptomatic at one year of follow-up.

Fig. 3.

Left: Intraoperative image showing cecal duplication cyst (red arrow), Right: operated specimen. (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)

Fig. 4.

Intraoperative image showing Meckel's diverticulum with wide base (white arrow).

3. Discussion

Mortality attributed to bleeding, perforation, and malignancy in EDC is linked to delays in diagnosis and subsequent treatment [10]. Differentiating EDC from MD is a significant challenge, as both can present with similar symptoms and be positive on a Tc-99 m radionuclide scan [11,12]. The final diagnosis of EDC and MD can only be made by correlating the imaging findings with the surgical results and pathological features [13,14]. The etiology of EDC remains uncertain. Several theories have been proposed, including the persistence of embryonic diverticula during git development, intrauterine vascular accidents, the split notochord theory and recanalization and fusion of embryonic longitudinal folds [15,16]. EDCs can manifest at any age, with 80 % of patients being diagnosed within the first two years of life, as in our case. The symptoms vary based on the type, size, morphology, location of the cysts and communication with the git [17]. Cecal duplications typically manifest with symptoms of acute intestinal obstruction, as in our case [18]. The mechanism of obstruction is the presence of mucus within the lumen. MD is a true diverticulum arising from the persistence of the omphalomesenteric duct. It can be asymptomatic or can present with gastrointestinal bleeding, diverticulitis, obstruction, or intussusception [19].

The preferred treatment for CDC is surgical resection and primary end-to-end anastomosis. Various surgical techniques have been proposed for this purpose [11]. Performing diverticulectomy for incidental MD during abdominal surgery is controversial. A systematic review indicated that diverticulectomy for incidentally detected MD resulted in a significantly greater postoperative complication rate than did drainage [20]. Resection of the MD was not performed in our patient because the MD was non-inflamed, without perforation and base of the MD was wide.

Due to the paucity of cases, there are no recommended guidelines for the treatment of coexisting EDC and MD. We opted for surgical removal of the CDC through resection followed by primary anastomosis. This approach is similar to that used in prior cases with coexisting cystic diverticula, MD and tubular diverticula, in which the ileal segment containing them was resected and ileo-ileal anastomosis was performed [21].

4. Conclusion

Cecal duplication cysts should be considered a differential diagnosis in infants who present with intestinal obstruction. Although the presentation may resemble that of Meckel's diverticulum, both conditions can coexist. Excision of duplication cysts in children is considered a safe and efficient treatment approach. The surgical approach should be individualized, with complete removal recommended whenever possible.

Consent

Written informed consent was obtained from the parents of the patient for publication and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal upon request.

Ethical approval

Since this is a case report, our Institutional Review Board waived the requirement for ethical approval.

Declaration of generative AI in scientific writing

Chat GPT 3.5 was used to improve readability and language; however, we assure that the authors are ultimately responsible for and accountable for the contents of the work.

Funding

This study received no funding.

Author contribution

Conceptualization: Santosh Dev, Prajjwol Luitel, Sujan Paudel, Badal Karki, Barsha Dev, Geha Raj Dahal.

Patient Management: Santosh Dev, Geha Raj Dahal.

Writing – original draft: Santosh Dev, Prajjwol Luitel, Sujan Paudel, Badal Karki, Barsha Dev, Geha Raj Dahal.

Writing – review & editing: Santosh Dev, Prajjwol Luitel, Sujan Paudel, Badal Karki, Barsha Dev, Geha Raj Dahal.

Visualization and Supervision: Santosh Dev, Prajjwol Luitel, Sujan Paudel, Badal Karki, Barsha Dev, Geha Raj Dahal.

Registration of research studies

N/A

Guarantor

Prajjwol Luitel

Conflict of interest statement

No conflicts of interest were identified in the preparation of this case report.

Data availability

The datasets used during this study will be available from the corresponding author upon reasonable request.