Abstract
Introduction and significance
Duplication of the digestive tract is a rare condition, especially when it is located in the cecum. In most cases, it is asymptomatic. The cystic pattern is the most common type of duplication of the digestive tract. The exact cause of this anomaly has not been determined yet.
Case presentation
We present the case of a girl who was suffering from vague abdominal pain for a period of time without response to medical treatment. She was later diagnosed with a duplication cyst of the cecum, which was completely resected with a ileocolic anastomosis.
Clinical discussion
Surgery is generally the mainstay of treatment for duplication cysts of the digestive tract. Even in asymptomatic cases, surgical resection is preferred to avoid complications and the risk of malignant transformation.
Conclusion
It is always important to consider duplication of the digestive tract as a possible differential diagnosis for cysts in the cecum, and to avoid attempting to dissect the cyst away from the adjacent digestive tract.
Keywords: Case report, Duplication, Cecum, Digestive tract, Pediatric surgery
Highlights
-
•
Intestinal duplication cysts are very rare in the cecum.
-
•
The primary radiological investigation is considered to be ultrasound.
-
•
Surgical treatment is considered to be the mainstay of intestinal duplication cysts.
-
•
It is possible that some malignant cases develop from intestinal duplication cysts.
1. Introduction
In general, duplication of the digestive tract is a condition that refers to the duplication of one of the parts of the digestive tract extending from the mouth to the anus [1].
The incidence of duplication is estimated to be approximately 1 in 4,500 live births, making it a rare condition [2].
Most cases present in children under the age of two years [3].
There are two types of duplication of the digestive tract: cystic and tubular, with most cases being cystic [4].
Several theories have been proposed to explain this condition, but there is no single theory that definitively explains the underlying cause.
The clinical presentation depends on the location and size of the duplication. Some cases may be asymptomatic and discovered incidentally.
Duplication of the cecum is one of the rarest locations for duplication of the digestive tract, accounting for approximately 0.4% of all cases of duplication of the digestive tract [5].
This case is described in accordance with the criteria of SCARE [6].
2. Presentation of case
2.1. Patient information
We describe the case of a 7-year-old girl who has been suffering from abdominal pain in the left iliac fossa and right lumbar region for about a week. There is no fever or anorexia, and the pain responds slightly to medication.
There are no other gastrointestinal symptoms such as vomiting, hematochezia, or diarrhea. The girl's family has no history of tumors, abdominal lesions, or malformations.
3. Clinical findings
Upon arrival of the girl in the emergency room, her general condition was good.
On physical examination, her vital signs were good with a soft abdomen without abdominal guarding, but a mass was palpated in the right iliac fossa and extended to the corresponding lumbar region with pain on palpation of the mass.
There were no signs of peritoneal irritation.
The rest of the physical examination was within normal limits.
4. Diagnostic assessment
Laboratory examinations L revealed a hemoglobin of 13.8 g/dL, total white blood cell count of 13,000/mm3, platelet count of 487,000/mm.
All tumor markers, including carcinoembryonic antigen (CEA), CA 125, CA19–9, AFP, and B HCG were within normal ranges.
Abdominal ultrasound showed the presence of a large cyst was observed filling the cecum and ascending colon without connection to the liver or right kidney.
The CT scan showed a large cystic mass is seen to lie lateral and superior to the cecum and ascending colon without evidence of connection to the right kidney or liver. (Fig. 1.A-B).
Fig. 1.
A: CT /cross coronal view showing a large cystic mass has formed on the adhesion of the cecum and ascending colon without connection to the liver, where the blue color indicates enteric duplication cyst
B: CT /cross axial view showing a cystic mass has formed in the right iliac fossa, attached to the cecum with compression of the cecum, where the yellow color indicates the cecum, the green color indicates enteric duplication cyst, (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)
5. Therapeutic intervention
Based on the previous findings, it was decided to perform surgery.
The procedure was performed at our tertiary college teaching hospital. It was performed by a pediatric surgery specialist.
Under general anesthesia, A transverse incision was made above and to the right of the umbilicus, extending to the abdomen. A cystic mass was found extending from the cecum to the middle of the ascending colon, with adhesions in the upper part of the cyst with a common wall between the cyst, cecum, and ascending colon (Fig. 2.A). It was decided to perform a complete resection of the cyst with the cecum and part of the ascending colon (Fig. 2.B), and perform a ileocolic anastomosis with the ascending colon and closure of the layers.
Fig. 2.
A: Intraoperative image, where the blue color indicates the cecum, the yellow color indicates enteric duplication cyst and the green color indicates appendix.
B: Image after complete excision of the cyst with cecum and appendix. (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)
Histopathologic examination shows an enteric duplication cyst in the cecum (Fig. 3.A-B).
Fig. 3.
A: Histopathological analysis of the resected cyst, was lined mainly by gastric and esophageal-type epithelium (H&E stain).
B: Histopathological analysis of the resected cyst, was lined by pseudostratified ciliated respiratory type epithelium with goblet cells (H&E stain).
The child was monitored in the hospital for 5 days and then discharged in good general condition.
6. Discussion
In the past, many terms were used to describe duplication of the digestive tract, such as intestinal cysts, giant diverticulum, or atypical Meckel's diverticulum. Lad later introduced the term “duplication of the digestive tract” to clarify these terms [7].
Duplication of the digestive tract is a microscopic structure that resembles the intestine, containing muscular layers and taking the form of hollow structures, either tubular or the other more common form, which is cystic. It is located near one of the parts of the digestive tract to which it is connected, where this connection can be with the muscular wall of the adjacent digestive part. It may also share the blood supply and be located on the mesenteric side of the adjacent digestive part [[8], [9]].
Duplication of the digestive tract can occur anywhere along the entire digestive tract, with the ileum being the most common location and duplication of the cecum being rare [10].
Depending on the location, size, and shape, duplication of the digestive tract can present with a variety of symptoms. Duplication in the anterior intestine may present with symptoms associated with respiratory symptoms, while duplication of the middle and posterior intestines may present with bleeding, obstructive symptoms, or perforation. Duplication may also present with vague abdominal pain, as occurred in our case.
Occasionally, the mucosa of the duplication may contain ectopic gastric tissue, which increases the risk of perforation or ulceration [11].
Duplication of the cecum may present like duplication of the digestive tract when it is located in the abdomen and may be the starting point for colonic intussusception [12].
In rare cases, duplication of the digestive tract may remain asymptomatic.
Many theories have attempted to find a clear explanation for the occurrence of duplication of the digestive tract to reach an accurate understanding of this condition, but no theory has been able to determine the exact cause of this rare anomaly. The theory of the retention of the diverticulum during embryonic development is the most widely accepted [13].
Cystic duplication of the cecum often appears as a mass in the abdomen, and the differential diagnosis of cystic lesions in the abdomen includes cecum masses, tumors, duplication cysts, dermoid cysts, and Meckel's diverticulum cysts [14].
As for radiological investigations, abdominal ultrasound is the ideal investigation, with a sensitivity of up to 95 % and a positive predictive value of 85–100 %. There is no need to perform a CT scan in all cases.
If there is a suspicion of ectopic gastric mucosa within the duplication, a Technetium pertechnetate scan can be performed to help identify the ectopic gastric mucosa [15].
Many congenital anomalies associated with duplication of the digestive tract may be helpful in diagnosis, such as congenital heart disease, congenital lung malformations, and meningocele with anterior intestinal malformations, while spinal cord malformations may be seen frequently with duplication of the anterior and posterior intestines.
Inversion of the bladder and imperforate anus may be seen with duplication of the posterior intestine [16].
The gold standard treatment for symptomatic intestinal duplication cysts is complete resection.
However, the treatment of asymptomatic duplication of the digestive tract is controversial, and complete resection is preferred due to the potential for serious complications and malignant transformation [17].
Malignant changes are a rare complication of duplication of the digestive tract. Several cases of malignant tumors arising from the small intestine and also as a development of duplication cysts in the large intestine have been reported [[18], [19]].
7. Conclusion
The presence of vague abdominal pain in a child warrants a serious study of the case and not continuing with drug treatment for a long time, in order to avoid exposing the child to the risks of complications of duplication cysts of the digestive tract, and to focus on the complete resection of the cysts during surgery.
Abbreviations
- US
ultrasound
- CT
Computed tomography
Funding
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Author contribution
M.J: Conceptualization, resources, who wrote, original drafted, edited, visualized, validated, literature reviewed the manuscript, and the corresponding author who submitted the paper for publication.
S.J and H.O: Supervision, visualization, validation, resources, and review of the manuscript.
H.J, M.T.M and M.H.: Visualization, validation, and review of the manuscript.
All authors read and approved the final manuscript.
Guarantor
Marwa Jaweesh.
Research registration number
N/A
Provenance and peer review
Not commissioned, externally peer-reviewed.
Consent of patient
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
Declaration of competing interest
The authors declare that they have no competing interests.
Contributor Information
Shkri Jaweesh, Email: shkri.jaweesh002@gmail.com.
Marwa Jaweesh, Email: marwajaweesh123@gmail.com.
Data availability
The datasets generated during and/or analyzed during the current study are not publicly available because the Data were obtained from the hospital computer-based in-house system. Data are available from the corresponding author upon reasonable request.
References
- 1.Macpherson R.I. Gastrointestinal tract duplications: clinical, pathologic, etiologic, and radiologic considerations. Radiographics. 1993;13:1063–1080. doi: 10.1148/radiographics.13.5.8210590. [DOI] [PubMed] [Google Scholar]
- 2.Puligandla P.S., Nguyen L.T., St-Vil D., Flageole H., Bensoussan A.L., Nguyen V.H., Laberge J.M. Gastrointestinal duplications. J. Pediatr. Surg. 2003;38(5):740–744. doi: 10.1016/jpsu.2003.50197. [DOI] [PubMed] [Google Scholar]
- 3.Liu R., Adler D.G. Duplication cysts: diagnosis, management, and the role of endoscopic ultrasound. Endoscopic ultrasound. 2014;3(3):152–160. doi: 10.4103/2303-9027.138783. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 4.Kanbar M., Alomar K., Alkader M.A., Alloun M., Al Dalati H., Alghazal L.K. A unique case of complete tubular colonic duplication with normal anus accompanied by vestibular fistula in a 12 years _ old _ girl - a case report. Int. J. Surg. Case Rep. 2023;108 doi: 10.1016/j.ijscr.2023.108405. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 5.Temiz A., Oğuzkurt P., Ezer S.S., İnce E., Gezer H.Ö., Hiçsönmez A. Different clinical presentations, diagnostic difficulties, and management of cecal duplication. J. Pediatr. Surg. 2013;48(3):550–554. doi: 10.1016/j.jpedsurg.2012.07.048. [DOI] [PubMed] [Google Scholar]
- 6.Sohrabi, C., Mathew, G., Maria, N., Kerwan, A., Franchi, T., Agha, R. A., & Collaborators (2023). The SCARE 2023 guideline: updating consensus surgical CAse REport (SCARE) guidelines. Int. J. Surg. (London, England), 109(5), 1136–1140. doi: 10.1097/JS9.0000000000000373. [DOI] [PMC free article] [PubMed]
- 7.Gastrointestinal Duplications: Practice Essentials, Anatomy, Pathophysiology. https://emedicine.medscape.com/article/936799-overview [Internet]. [cited 2022 Oct 10]. Available from:
- 8.Gross R.E., Holcomb G.W., Jr., Farber, S. Duplications of the alimentary tract. Pediatrics. 1952;9(4):448–468. [PubMed] [Google Scholar]
- 9.Al-Nahar L.A., Kafaween H.M., Mohaidat T.S. Cecal enterogenous duplication cyst: a case report in an adult. JRMS. 2011;18:58–60. [Google Scholar]
- 10.Radhakrishna V., Rijhwani A., Jadhav B. 2018. Cecal duplication: a mimicker of intussusception: a case report and review; pp. 17–19.https://www.sciencedirect.com/science/article/pii/S2049080118301134 Available from, Ann. Med. Surg. [Internet]. 31. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 11.Srikanth K.P., Thapa B.R., Lal S.B., Menon P., Sodhi K., Vaiphei K., Rao K.L. Non communicating gastric antral duplication cyst presenting with hematemesis due to large antral ulcer. Trop. Gastroenterol. 2015;36(2):134–136. [PubMed] [Google Scholar]
- 12.Alomar K., Alhariri A., Alloun M., Dawarah M., Alkader M.A., Qatleesh S. A unique case of enteric duplication cyst in the cecum presenting as ileocolic intussusception in a 3-year-old child: case report and review of the literature. Int. J. Surg. Case Rep. 2023;112 doi: 10.1016/j.ijscr.2023.108935. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 13.Mourra N., Chafai N., Bessoud B., Reveri V., Werbrouck A., Tiret E. Colorectal duplication in adults: report of seven cases and review of the literature. J. Clin. Pathol. 2010;63(12):1080–1083. doi: 10.1136/jcp.2010.083238. [DOI] [PubMed] [Google Scholar]
- 14.Tiwari C., Shah H., Waghmare M., Makhija D., Khedkar K. Cysts of gastrointestinal origin in children: varied presentation. Pediatric gastroenterology, hepatology & nutrition. 2017;20(2):94–99. doi: 10.5223/pghn.2017.20.2.94. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 15.Pati A., Mohanty H.M., Subudhi P.C., Dash R., Mohanty P.K., R.K. Mahapatra duplication cyst of the caecum: a case report. Indian J. Surg. 2010;72:271–272. doi: 10.1007/s12262-010-0075-7. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 16.Puri P., Hollwarth M.E. Springer Publishing; 2019. Pediatric Surgery. [Google Scholar]
- 17.Hata H., Hiraoka N., Ojima H., Shimada K., Kosuge T., Shimoda T. Carcinoid tumor arising in a duplication cyst of the duodenum. Pathol. Int. 2006;56(5):272–278. doi: 10.1111/j.1440-1827.2006.01957.x. [DOI] [PubMed] [Google Scholar]
- 18.Hsu H., Gueng M.K., Tseng Y.H., et al. Adenocarcinoma arising from colonic duplication cyst with metastasis to omentum: a case report. J. Clin. Ultrasound. 2011;39:41–43. doi: 10.1002/jcu.20739. [PubMed] [Google Scholar] [DOI] [PubMed] [Google Scholar]
- 19.Takrouny and Abdelmohsen 2020 had Published 2 Cases Like Your Case Report doi: 10.1016/j.epsc.2020.101499. [CrossRef]. [DOI]
Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Data Availability Statement
The datasets generated during and/or analyzed during the current study are not publicly available because the Data were obtained from the hospital computer-based in-house system. Data are available from the corresponding author upon reasonable request.