|Year : 2016 | Volume
| Issue : 2 | Page : 115-117
Cecal duplication cyst: A rare case report with review of literature
Vikas Shyam Raj Singh, Prasmit Amit Shah, Prakash M Roplekar, S Sudhamani, Prathmesh Desai
Department of Pathology, DY Patil University School of Medicine, Navi Mumbai, Maharashtra, India
|Date of Web Publication||14-Apr-2016|
Vikas Shyam Raj Singh
Department of Pathology, DY Patil University School of Medicine, Nerul, Navi Mumbai - 400 706, Maharashtra
Source of Support: None, Conflict of Interest: None
Duplication cysts of the alimentary tract are very rare congenital anomalies. Out of all these cases, two-thirds of them manifest before the age of 2 years. They are common in ileum, but very rare in cecum. Some of them may remain asymptomatic and present in the adulthood. The lesion may be tubular or cystic. Several theories have been postulated, but true etiology is not known. We hereby report a case of a 10-year-old female who presented with abdominal pain in the pediatric surgery outpatient department. Diagnosis of cecal duplication cyst was confirmed on histopathology. This report implies that although alimentary tract duplications are rare, they should be considered in the differential diagnosis of children who presents with acute abdominal pain.
Keywords: Cecum, duplication cyst, intussusception
|How to cite this article:|
Singh VS, Shah PA, Roplekar PM, Sudhamani S, Desai P. Cecal duplication cyst: A rare case report with review of literature. Int J Health Allied Sci 2016;5:115-7
|How to cite this URL:|
Singh VS, Shah PA, Roplekar PM, Sudhamani S, Desai P. Cecal duplication cyst: A rare case report with review of literature. Int J Health Allied Sci [serial online] 2016 [cited 2021 Dec 1];5:115-7. Available from: https://www.ijhas.in/text.asp?2016/5/2/115/180425
| Introduction|| |
Gray remarked, “gastrointestinal tract is a fertile field for various and curious congenital malformations, fascinating to study for both surgeon and pathologists.” Duplications of the alimentary tract are very rare malformations which vary in size, appearance, location, and symptoms. Multiple theories have been proposed, but no single theory explains the all known variants. It can occur in any part of the gastrointestinal tract from esophagus to anus and always occurs on the mesenteric side. Majority occur in ileum, but are very rare in cecum. The incidence is around 1 in 4500 births. Majority of them are present in the first 2 years of life. Until now, only 19 cases of cecal duplication cyst have been reported in the English literature. Duplication cysts are often confused as intussusception, appendicitis, or Meckel's diverticulum. Surgery is the treatment of choice and this condition has an excellent prognosis.
| Case Report|| |
A 10-year-old female, with no history of chronic illness came to Pediatric Surgery Outpatient Department with complaints of pain in the right iliac fossa, vomiting, and abdominal distension. There was no history of fever or diarrhea. On examination, abdominal tenderness was noted mainly in the right iliac fossa. Palpable and movable lump was felt in the infraumbilical region measuring 3 cm × 3 cm. Clinical diagnosis was acute intestinal obstruction with a possibility of intussusception.
Laboratory findings showed complete blood count parameters and serum electrolytes in normal range. Beta human chorionic gonadotropin levels were <0.05 mIU (normal range - <5 mIU). Abdominal and pelvic ultrasonography revealed a cystic mass measuring 4 cm × 4 cm in the right iliac fossa with the presence of intraluminal fluid in the cyst and signs of intestinal obstruction. Contrast-enhanced computed tomography (CT) scan confirmed the cystic mass without septations at the ileocecal junction. Exploratory laparotomy was done with midline incision on the lower abdomen. Intraoperatively, a cystic mass was noted in the cecum. No intussusception was noted on exploration. Gradual dissection of the cyst was performed. The lesion was resected with end-to-end anastomosis.
An already cut opened segment of ileum, cecum with attached appendix and part of ascending colon altogether measuring 10 cm × 7 cm × 1.5 cm, was sent for the histopathological examination. A cystic structure is seen at cecum mesenteric border measuring 3.5 cm × 3.3 cm. Ileum, appendix, and part of ascending colon were unremarkable. No areas of necrosis or hemorrhage were noted [Figure 1].
Microscopic examination of the cecal cyst showed all layers of large intestine displaying mucosa with diffuse chronic inflammatory infiltrate and prominent lymphoid follicles in the submucosa. Muscularis propria is thickened with congested and dilated blood vessels. There was no evidence of ectopic or abnormal tissue [Figure 2]. Appendix showed features of recurrent appendicitis. Rest of the mucosa of ileum and part of the ascending colon are unremarkable. The child was followed up for 6 months and is apparently normal.
|Figure 2: H and E, ×4 from the cystic lesion of cecum showing all layers of large intestine|
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| Discussion|| |
The congenital intraabdominal cysts are classified as lymphatic or chylous cysts, enterogenous cysts, urogenital cysts, dermoid cysts, or terotoids. They are rare congenital anomalies that can occur anywhere in the gastrointestinal tract from mouth to anus on the mesenteric side.
This case was first reported by Coider in 1733. William E Ladd in 1937 used the term “duplications of alimentary tract.” In 1941, Ladd and Gross reported 18 such cases while in 1953 Gross reported 68 such cases. A meta-analysis was carried out by Heiss encompassing 580 patients, demonstrated that 20% of such lesions occur in the chest whereas 80% occur in the abdomen.
Colonic duplications are rare (13%). Cecal duplications are even rarer as only >19 cases have been reported in the English literature. Eighty percentage of these cases present in the first 2 years of life, but it has also been reported in adults. The most frequent location for a duplication cyst of the intestinal tract is the region of terminal ileum and ileocecal valve. Next to it are esophagus, stomach, and duodenum. Cecum and colon are the rarest sites, the incidence being 13%.
In an 18-year review, Grosfield et al. could find only one case of cecal duplication in the neonate. Oudshoom and Heij reviewed around 368 cases and found that only 16 of them were cecal duplication cysts. The clinical and radiological preoperative diagnosis of the duplication cyst is difficult due to variation in the signs and symptoms. These symptoms include abdominal distension, vomiting, palpable abdominal mass, bleeding, and rarely urinary hesitancy.
They are also associated with other congenital malformations such as vertebral and urogenital abnormalities. Complications include perforation, intussusception, bowel obstruction, volvulus, and associated malignancy.
The most common imaging modalities to diagnose duplication cysts are ultrasonography and Barium studies whereas CT and magnetic resonance imaging (MRI) are less often used. But, nowadays, diagnostic laparoscopy is widely used. Ultrasonography shows characteristic echogenic inner mucosal layer and hyperechoic outer muscular layer. Barium study reveals submucosal mass extending up to the lumen of the gastrointestinal tract. CT scan reveals smooth, rounded, fluid-filled cysts with thin, slightly enhancing walls. MRI scan will show intracystic fluid with heterogeneous signal density on the T1-weighted images and homogenous high signal intensity on the T2-weighted images.
Pathological evaluation of the enteric cysts is the mainstay of diagnosis. Gross and microscopic appearance showing mucosa, submucosa, muscularis, and serosa are the typical features. Good sectioning of the cyst wall with the attached bowel helps in ruling out the malignant changes.
Various surgical procedures have been employed to deal with such lesions. Most commonly used is the partial colectomy with end-to-end anastomosis. Other techniques such as enucleation, marsupialization, or evacuation of cyst can also be used.
| Conclusion|| |
Duplication cysts are to be considered in the differential diagnosis in neonates and children who present with acute pain and palpable abdominal mass.
Whereas the ultrasound is the best preoperative diagnostic tool and thorough histopathological examination is the best confirmative method of diagnosis. Resection is the treatment of choice with an excellent outcome.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Zahir I, Yusuf S, Zada F, Asif M, Akhtar N, Abbasi MZ. Duplication cyst in a new born. Int J Pathol 2010;8:84-6.
Demir N, Melek M, Kaba S, Zehra Dogan S, Tuncer O. Duplication cysts of the cecum and duodenum in an infant: A rare case report. New J Med 2014;31:137-8.
Al-Nahar LA, Kafaween HM, Mohaidat TS. Cecal enterogenous duplication cyst: A case report in an adult. JRMS 2011;18:58-60.
Lahoti HN, Singh RV. Ileo-cecal duplication cyst masquerading as intussusception. J Case Rep 2013;3:410-2.
Sudhamani S, Nahar A, Pandit A, Kiri VM. Duplication cyst of the caecum – A rare case report with review of literature. Bombay Hosp J 2011;58:82-4.
Ijaz L, Husnain M, Malik SI, Mirza B. Cecal duplication cyst presenting as acute intestinal obstruction in an infant. APSP J Case Rep 2011;2:11.
[Figure 1], [Figure 2]