• Users Online: 129
  • Print this page
  • Email this page


 
 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 12  |  Issue : 2  |  Page : 78-80

Oesophageal duplication – An uncommon cause of dysphagia in an adult


Department of General Surgery, Maulana Azad Medical College and Associated Hospitals, New Delhi, India

Date of Submission11-Jul-2021
Date of Decision20-Nov-2021
Date of Acceptance22-Dec-2021
Date of Web Publication26-Apr-2022

Correspondence Address:
Dr. Rajdeep Singh
Department of General Surgery, 2nd Floor, B. L. Taneja Block, Maulana Azad Medical College Complex, New Delhi - 110 002
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/cmrp.cmrp_68_21

Rights and Permissions
  Abstract 


Duplication of the oesophagus has ranges from complete duplication which is very rare to a partial cystic duplication (also known as duplication cyst). The oesophagus is the second most common site after the ileum to be involved with gastrointestinal duplication. It commonly manifests in neonates and children. The adults are usually asymptomatic. In this report, we describe a case of an adult male presenting with dysphagia because of a lower oesophageal duplication cyst. The diagnostic features and treatment options are discussed.

Keywords: Oesophageal duplication, oesophageal duplication cyst, congenital foregut duplication, dysphagia in adult


How to cite this article:
Singh A, Singh R, Mishra A, Agarwal PN. Oesophageal duplication – An uncommon cause of dysphagia in an adult. Curr Med Res Pract 2022;12:78-80

How to cite this URL:
Singh A, Singh R, Mishra A, Agarwal PN. Oesophageal duplication – An uncommon cause of dysphagia in an adult. Curr Med Res Pract [serial online] 2022 [cited 2022 May 19];12:78-80. Available from: http://www.cmrpjournal.org/text.asp?2022/12/2/78/343936




  Introduction Top


A duplication cyst of the oesophagus is a type of congenital foregut duplication. It is an uncommon condition, with an estimated incidence of 1 in 100,000 in children. In most cases, up to 80% are diagnosed during childhood; it is rare in adults, with approximately 160 cases reported till 2014.[1] A reported incidence of 1 in 8200 is seen among autopsies.[2] They comprise 10%–25% of all duplication cysts of the gastrointestinal tract, with ileal duplication being the most common.[3] Males are more commonly affected.[4] The most common site is the lower third (60%), followed by the upper and middle third (20% each).[5]

We report a case of oesophageal duplication cyst in an adult, presenting with dysphagia and its management.


  Case Report Top


A 52-year-old diabetic male presented with dysphagia to solid food and postprandial epigastric and retrosternal discomfort for the past 2 months. The general and systemic examination was unremarkable, apart from a small umbilical hernia. All routine investigations were normal. Chest X-ray showed a round-to-oval homogeneous radiopacity in the left cardiac region with an ill-defined lower margin [Figure 1]a. On endoscopy, a smooth swelling was present on the left lateral wall of the gastroesophageal junction, with normal mucosa [Figure 1]b, suggestive of extrinsic compression. A barium swallow had similar findings [Figure 1]c. Contrast-enhanced computed tomography (CECT) showed a thick-walled cystic lesion measuring 6.5 cm × 5.3 cm × 4.9 cm, at the distal left paraesophageal region. Air focus was seen within the cyst; however, oral contrast did not enter the cyst [Figure 1]d.
Figure 1: (a) Chest radiograph, with smooth opacity in left cardiac region. (b) Barium swallow showing a smooth outline. (c) Computed tomography showing a smooth-walled, cystic lesion in relation to distal oesophagus (d) Endoscopic view of a smooth-walled extrinsic compression

Click here to view


Through an upper midline abdominal incision, the oesophagus was mobilised by preserving vagus nerves. The hiatus was enlarged by dividing crural fibres, and the cyst was exposed. Adhesions with the left lung were separated, and the cyst could be separated from all around except at the oesophagus. The cyst was opened, thick whitish fluid was evacuated and the wall excised until the junction of the cyst with the oesophagus [Figure 2]. No luminal continuity was seen after opening the cyst. The mucosa of the cyst overlying oesophagus was removed, but no attempt was made to separate the tightly adherent cyst wall from the oesophagus. Omentum was loosely placed over the raw area, so created and was secured with a single stitch. After confirming haemostasis, a chest tube was placed and hiatus was repaired. The abdomen was closed conventionally.

Dysphagia was relieved from the first post-operative day. Histopathological examination revealed extensive areas of mucosal ulceration with dense acute on chronic inflammatory granulation tissue and cholesterol clefts. Foci of antral metaplasia were also seen. The muscular layer was identified in the cyst wall and thus was consistent with a diagnosis of true duplication cyst with metaplastic changes [Figure 3].
Figure 2: Showing part of cyst wall excised

Click here to view
Figure 3: Histopathology specimen showing muscle layers

Click here to view


The patient was discharged with the resolution of symptoms in the post-operative period and did not suffer any complications of surgery. The patient on the follow-up visit has no recurrence of symptoms.


  Discussion Top


Embryologically, oesophageal duplication cyst is a type of congenital foregut duplication, with duplication of the muscularis mucosa and externa without epithelial duplication. This variant is more common as compared to complete duplication of the oesophagus.[6] For classifying a foregut cyst as an oesophageal duplication cyst, the following criteria need to be met: (1) the cyst must be within or attached to the oesophageal wall, (2) it must be covered by two muscle layers and (3) the lining must consist of squamous, columnar, cuboidal, pseudostratified or ciliated epithelium.[2] Most duplication cysts are reported in the lower oesophagus, with only 23% occurring in the upper third.[5]

Clinical presentation varies, with the most common symptom being dysphagia, epigastric discomfort and retrosternal pain in adults. The majority of patients develop symptoms during adolescence, whereas symptomatic cysts are rarely encountered in adulthood (<7%), and the diagnosis is usually suspected from an incidental finding on a chest radiograph. Chronic cough, recurrent respiratory infections or tracheal compression are more common with upper oesophageal cysts and usually manifest in early childhood.[1],[7] Karaman et al. reported a rare case that presented with back pain.[3] Acute onset is due to complications by intracystic haemorrhage, perforation and infection, especially in those with oesophageal communication.[7] Malignant transformation into adenocarcinoma and squamous cell carcinoma has also been reported, though extremely rare.[1],[4],[7]

Diagnosis is often made from an incidental finding on the chest radiograph; however, contrast computed tomography and endoscopic ultrasound (EUS) are diagnostic. A barium swallow will rule out mucosal disease, showing a smooth extrinsic compression. Computed tomography demonstrates the cystic nature of the mass, along with perforation if present. It also helps by ruling out other causes, by evaluation of the spine, pulmonary parenchyma, airway and adjacent structures.[8] Diagnosis may also be obtained by EUS, although it is less helpful in ruling out a bronchogenic cyst. EUS has an advantage over CECT as it can be both diagnostic as well as therapeutic. This is because the cyst content can be aspirated for immediate temporary relief of symptoms and for the histological analysis of the cyst content.[8],[9]

Differential diagnosis of a posterior mediastinal cyst includes neurogenic tumour, bronchogenic cyst, epiphrenic diverticulum, pericardial cyst and cystic lymphangioma/hygroma. In endemic countries, a hydatid cyst of the lung can be confused with thoracic oesophageal duplication and a hydatid cyst of the liver with those of the abdominal oesophagus.[3]

Surgical resection through posterolateral thoracotomy incision has been considered the best surgical approach in symptomatic cases. This has been replaced by video-assisted thoracoscopic surgery (VATS), which reduces post-operative pain, leads to earlier recovery and has a better cosmetic outcome. In this case, we used an abdominal approach because our differential diagnosis included epiphrenic diverticula, which would have required Heller's myotomy and fundoplication, which was considered to be easier by this approach.[8],[9],[10]

If available, robotic assistance may be used. Compared with VATS, robotic has a few advantages: (1) superior visualisation of the surgical field, (2) greater range of motion with multi-articulated instruments and (3) more precise movements with tremor filtration and motion scaling. Obasi et al. presented two paediatric patients who underwent successful surgical resection of oesophageal duplication cysts through robotic VATS. They had no operative complications with excellent post-operative outcomes including decreased pain and early patient discharge.[11]

As the true prevalence of oesophageal cysts is unknown, it is very likely that many adults have no symptoms, remain undetected and require no intervention.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Bassem N, Fethi D, Mohamed BM, Jaafar M, Waad F, Malek B, et al. Complicated esophageal duplication cyst in adult: A case report and review of the litterature. J Gasteroenterol Hepatol Res 2015;4:1801-4.  Back to cited text no. 1
    
2.
Arbona JL, Fazzi JG, Mayoral J. Congenital esophageal cysts: Case report and review of literature. Am J Gastroenterol 1984;79:177-82.  Back to cited text no. 2
    
3.
Karaman I, Gürel D, Ünlü M, Yilmaz E, Karaçam V, Kargi A. Esophageal duplication cyst: A rare cause of back pain. Turk J Gastroenterol 2012;23:610-1.  Back to cited text no. 3
    
4.
Jacob R, Hawkes ND, Dallimore N, Butchart EG, Thomas GA, Maughan TS. Case report: Squamous carcinoma in an oesophageal foregut cyst. Br J Radiol 2003;76:343-6.  Back to cited text no. 4
    
5.
Sodhi KS, Saxena AK, Narasimha Rao KL, Singh M, Suri S. Esophageal duplication cyst: An unusual cause of respiratory distress in infants. Pediatr Emerg Care 2005;21:854-6.  Back to cited text no. 5
    
6.
Eichmann D, Engler S, Oldigs HD, Schroeder H, Partsch CJ. Radiological case of the month. Denouement and discussion: Congenital esophageal duplication cyst as a rare cause of neonatal progressive stridor. Arch Pediatr Adolesc Med 2001;155:1067-8.  Back to cited text no. 6
    
7.
Pisello F, Geraci G, Arnone E, Sciuto A, Modica G, Sciumè C. Acute onset of esophageal duplication cyst in adult. Case report. G Chir 2009;30:17-20.  Back to cited text no. 7
    
8.
Cioffi U, Bonavina L, De Simone M, Santambrogio L, Pavoni G, Testori A, et al. Presentation and surgical management of bronchogenic and esophageal duplication cysts in adults. Chest 1998;113:1492-6.  Back to cited text no. 8
    
9.
Lim LL, Ho KY, Goh PM. Preoperative diagnosis of a paraesophageal bronchogenic cyst using endosonography. Ann Thorac Surg 2002;73:633-5.  Back to cited text no. 9
    
10.
Tan YK, Hui MT, Wong J, Yeo CT, Sng I, Ong YY. Four cases of developmental foregut cysts. Ann Acad Med Singap 1996;25:763-8.  Back to cited text no. 10
    
11.
Obasi PC, Hebra A, Varela JC. Excision of esophageal duplication cysts with robotic-assisted thoracoscopic surgery. JSLS 2011;15:244-7.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Discussion
References
Article Figures

 Article Access Statistics
    Viewed192    
    Printed0    
    Emailed0    
    PDF Downloaded12    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]