A 12-year-old boy presented with 2 episodes of large-volume hematochezia. He had undergone an appendicectomy at 5 months of age. Investigations revealed a hemoglobin of 7gm/dL, platelet count of 344000cells/cumm, partial thromboplastin time of 21.3 s, prothrombin time of 12.2 s, and international normalised ratio of 1.05. Colonoscopy showed a large sessile polyp(>2 cm) in caecum, multiple small(5-20 mm) sessile polyps in ileum and non-visualization of the appendicular opening(Fig. 1A). The caecal polyp was endoscopically unresectable. MR enterography showed T2-hypointense non-enhancing filling defect indenting anterior wall of caecum. He underwent exploratory laparotomy which revealed a caecal mass and Meckel’s diverticulum(MD), for which a right quarter colectomy (involving caecal mass and the ileo-caecal valve) with diverticular resection was done along with ileo-ileal anastomosis and ileo-ascending colon anastomosis. Gross examination revealed an ileo-caecal mass of 5x4x4cm(40gm) and small appendicular stump. Histopathology showed an enteric duplication cyst(EDC) lacking a muscle layer, lined by intestinal epithelium(Fig. 1B). Ileal segment containing the diverticulum showed gastric epithelium compatible with MD.
Figure 1.
(A) Colonoscopic image of the caecal polyp. (B) Duplication cyst- intestinal epithelium sharing a common submucosal layer.
EDC are rare congenital malformations with an incidence of 1:4500 births [1]. Common presentations include abdominal pain, abdominal mass or intussusception [2]. Recurrent hematochezia as a presenting feature of EDC has been rarely reported with a single case in adult woman. As in our patient, her ileo-colonoscopy showed a suspicious polyp-like lesion, which was surgically resected and identified to be an EDC [3]. Additionally, the co-occurrence of EDC and MD has been rarely reported, with patients presenting with abdominal pain or intestinal obstruction [4, 5]. Both can present similarly and have positive TC-99 m radionucleotide scans, thus, differentiating between them is done on the basis of radiological, surgical and histopathological findings [4]. In our patient, the absence of the appendicular opening and the presence of a caecal mass raised our suspicion, which led us to perform surgical removal and arrive at the diagnosis of a duplication cyst.
Acknowledgements
No form of financial or non-financial support has been received for this work.
Contributor Information
L K Pragathi, Department of Pediatric Gastroenterology and Hepatology, Bai Jerbai Wadia Hospital for Children, Mumbai, Maharashtra 400012, India.
Vishrutha Poojari, Department of Pediatric Gastroenterology and Hepatology, Bai Jerbai Wadia Hospital for Children, Mumbai, Maharashtra 400012, India.
Dhruv Gandhi, Department of Pediatric Gastroenterology and Hepatology, Bai Jerbai Wadia Hospital for Children, Mumbai, Maharashtra 400012, India.
Ira Shah, Department of Pediatric Gastroenterology and Hepatology, Bai Jerbai Wadia Hospital for Children, Mumbai, Maharashtra 400012, India.
Leena Patwardhan, Department of Pathology, Bai Jerbai Wadia Hospital for Children, Mumbai, Maharashtra 400012, India.
Pradnya Bendre, Department of Pediatric Surgery, Bai Jerbai Wadia Hospital for Children, Mumbai, Maharashtra 400012, India.
Author contributions
PLK, VP and DG wrote the manuscript draft. LP provided the histopathological images and reports for the draft. IS and PB critically reviewed and edited the manuscript. IS is the guarantor of this paper. All co-authors reviewed and approved the final draft.
Conflict of interest
The authors have no conflicting interests to declare regarding this manuscript.
Funding
No funding was received for this work.
Ethical approval
Not applicable.
Consent
Informed patient consent was obtained from the parent of the child.
Guarantor
Ira Shah is the guarantor for this manuscript.
References
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