Cecal duplication cyst in an adult: A case report

INTRODUCTION

Duplication cysts of the gastrointestinal tract are rare and cecal duplication cysts are rarer. Cecal duplication cysts are most commonly present within the first 2 years of life, but some rare cases may remain asymptomatic during childhood and may present in adult life.^[1] The clinical picture may vary according to the location, size, and type of the cyst. The most common symptoms of duplication cysts are that of an acute abdomen and are, therefore, very difficult to diagnose clinically.

In this article, we present an infrequent case of a cecal duplication cyst, which is itself rare, and presenting in adulthood is among the rarest phenomena. We address that the index of suspicion for a rare entity of duplication cyst should be kept in mind when the proximal loops are not dilated, but the patient has clinical and radiological signs of intestinal obstruction.

CASE REPORT

This is a case of 50-year-old female who presented to the emergency department of our hospital in September 2023 with chief complaints of palpable abdominal lump and progressive abdominal distension, abdominal pain in the right iliac fossa region. Along with this, the patient had symptoms of constipation, vomiting, abdominal fullness, tenderness, and palpitations. The patient had a history of constipation and post-prandial abdominal fullness and nausea for the last 15–20 years, for which she was taking medications for symptomatic relief. She has also been hospitalized several times for weakness, constipation, and nausea in the last 20 years. There are no comorbidities and significant family history associated.

On physical examination, the pulse rate was 120/min, blood pressure was 90/60 mmHg, temperature was 38°C, respiratory rate was 20/min, and oxygen saturation was 99% in room air. On systemic examination, there was abdominal distension, tenderness in the right lumbar region and right iliac fossa, and palpable abdominal mass in the right-sided abdominal region. Blood investigations revealed a total white blood cell count of 8,610/cu mm with mild neutrophilia, lymphocytosis and monocytosis, and hemoglobin of 11.5 g/dL. Radiograph of the abdomen in an erect position showed no significant abnormality. Ultrasound of the abdomen showed a gaseous abdomen. Bowel obstruction could not be ruled out. Contrast-enhanced computed tomography (CT) of the abdomen: Grossly dilated cecum measuring approximately 20 cm in transverse diameter extending up to the right dome of the diaphragm, the possibility of cecal volvulus. Other differentials: Diverticulum/duplication cyst [Figure 1].

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Figure 1:

3D multi-planar reconstruction of computed tomography images on oblique sagittal, coronal, and axial plane with labelings. (a) sagittal view - top white arrow: colonic diverticulum, middle white arrow: fecal matter filled cyst, bottom white arrow: caecum. (b) Coronal view - top yellow arrow: fecal matter filled cyst, bottom yellow arrow: Caecum. (c) Axial view: white arrow - fecal matter filled cyst.

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As the patient was diagnosed with cecal volvulus, she was taken for emergency exploratory laparotomy. Intraoperatively, a large cecal diverticulum with blind-ended huge colonic fecal matter-filled cystic lesion was identified for which resection of a huge diverticulum with ileocolic resection and ileotransverse anastomosis was done [Figure 2]. The patient was hemodynamically stable after the surgery. The patient was discharged from the hospital on the 7^th postoperative day without any remarkable post-operative events. Histopathological examination revealed features suggestive of cystic duplication of the colon without any granulomatous, inflammatory bowel disease, or malignant pathology. On follow-up visit after one month, the patient is quite happy and satisfied now.

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Figure 2:

Specimen after ileocecal resection showing a blind-ended fecal matter-filled cyst. top yellow arrow: ceacum, middle yellow arrow: colonic diverticulum, bottom yellow arrow: feacal matter filled cyst.

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DISCUSSION

Gastrointestinal duplication cysts are rare congenital anomalies and may occur in any part of the esophagus to the anus and are attached to the mesenteric side of the bowel.^[2]

Enteric duplication cysts are true diverticula of the bowel. They comprise an enteric mucosa, a muscle layer, and a nervous plexus.^[3]

Duplication cysts are embryological aberrations that are believed to occur between the 4^th and 8^th weeks of gestation, although the exact cause is not known. The following theories have been proposed to explain its etiopathogenesis: According to the split notochord theory, duplication cysts are abnormal separation of the growing notochord from the endodermal cells. This theory explains the association of vertebral anomalies with the duplication cysts.^[4] According to the recanalization theory, duplication cysts occur due to errors in the recanalization of the gut. According to the embryonic diverticula theory, a preexisting diverticula in the embryo grows with the growth of the alimentary tract which results in the development of duplication cysts. This theory is quite justifiable but is unable to explain the presence of heterotopic mucosa in these cysts.^[4,5]

The duplication cysts can be diagnosed by ultrasonography (USG), CT, magnetic resonance imaging, and technetium pertechnate scan. The double wall sign in USG has high specificity and positive predictive value still, it is quite subjective and difficult to distinguish from intussusception, volvulus, etc.^[6] However, CT remains the most effective modality to diagnose duplication cysts. In our case, it was difficult to distinguish between cecal volvulus and cecal duplication cyst. Surgical removal is the mainstay of treatment. Resection of the involved segment followed by end-to-end anastomosis is preferred for small cysts. For the larger cysts, where the segment involved is extensive and tubular, procedures such as enucleation, marsupialization, and excision of the mucosal layer are preferred to preserve the bowel length.^[7-9] Even in our case, the cyst was resected and then ileocecal anastomosis was preferred. Even after huge advancements in ultrasound imaging, only 20–30% of cases are being diagnosed antenatally.

DIFFERENTIAL DIAGNOSIS

Differential diagnoses of cecal duplication cyst can be cecal volvulus, meckel’s diverticulum, and intussusception. The distinguishing features are described in Table 1.

CONCLUSION

Cecal duplication cysts are rare congenital anomalies and difficult to diagnose as they present variably and have multiple differential diagnoses. Therefore, it is important to consider it in differentials when the patient presents with symptoms of acute abdomen. Although radiological investigations such as USG and CT are quite reliable, confirmatory diagnosis is made by histopathological examination.

TEACHING POINTS

1. Cecal duplication cysts commonly present early in life, but in some rare cases, they can even present later, and thus, it is important to consider it in differentials of acute abdomen.

2. Although the histopathological examination is best for confirmation of diagnosis, radiological modalities help in narrowing down the differentials and thus help in planning the line of management preoperatively.

MCQs

1. Which is the most effective radiological modality to diagnose a cecal duplication cyst?

   1. CT

   2. Ultrasonography

   3. Radiography

   4. MRI

   Answer Key: a

2. Which is the most common site for enteric duplication cysts?

   1. Cecum

   2. Rectum

   3. Ileum

   4. Colon

   Answer Key: c

3. Which is the gold standard investigation to diagnose a cecal duplication cyst?

   1. CT

   2. MRI

   3. Histopathological examination

   4. Ultrasonography

   Answer Key: c