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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 3  |  Issue : 1  |  Page : 20-22

Differentiation of esophageal duplication cysts from esophageal bronchogenic cysts: A case report and literature review


Department of General Surgery, People's Hospital of Dejiang, Tongren, Guizhou, China

Date of Submission18-Jan-2018
Date of Acceptance18-Feb-2018
Date of Web Publication22-Mar-2018

Correspondence Address:
Tao Zhou
Department of General Surgery, People's Hospital of Dejiang, 29 Mid Zhongshan RD, Dejiang, Tongren 565200, Guizhou
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ts.ts_1_18

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  Abstract 


Esophageal bronchogenic cysts located in inferior esophagus are rare. Here, we report on the case of a 12-year-old female patient who presented at our institution with dyspepsia and abdominal distention for 2 years with no other symptoms. Physical examination of abdomen and chest was negative, and laboratory tests did not show any abnormal results. However, computed tomography imaging showed a mass of about 8 cm × 8 cm × 4 cm in the lower esophagus compressing the stomach. The preoperative diagnosis was determined to be an inferior esophageal duplication cyst. A transabdominal excision of the cyst was performed 5 days after hospital admission. Pathology revealed an esophageal bronchogenic cyst. The case report and literature shows that differences exist between esophageal duplication cysts and esophageal bronchogenic cysts.

Keywords: Case report, Esophageal bronchogenic cyst, esophageal duplication cyst


How to cite this article:
Yang J, Zhou T, Fu J, Zhang Y. Differentiation of esophageal duplication cysts from esophageal bronchogenic cysts: A case report and literature review. Transl Surg 2018;3:20-2

How to cite this URL:
Yang J, Zhou T, Fu J, Zhang Y. Differentiation of esophageal duplication cysts from esophageal bronchogenic cysts: A case report and literature review. Transl Surg [serial online] 2018 [cited 2018 Apr 25];3:20-2. Available from: http://www.translsurg.com/text.asp?2018/3/1/20/228308




  Introduction Top


Bronchogenic cysts are extremely rare foregut abnormalities that arise from aberrant budding of the tracheobronchial tree early in embryological development.[1] They are often confused with cystic teratomas, bronchopulmonary sequestrations, cysts of urothelial, esophageal duplication cysts, and other foregut cysts.[2] These cysts are mostly located in the middle and superior mediastinum.[3] The esophageal duplication cyst has the same formation mechanism.[4] Both of these cysts may be adherent to the esophagus or intramural esophagus. Bronchogenic cysts are covered with nonkeratinizing squamous or ciliated columnar epithelium. However, esophageal duplication cyst walls contain two layers of smooth muscle accompanied by a lack of cartilage in the cyst wall. This information can be used to differentiate esophageal cysts from bronchogenic cysts.[5] However, such differentiation requires pathology information.


  Case Report Top


A 12-year-old female patient visited our hospital with a clinical presentation of dyspepsia and abdominal distention of the epigastrium ongoing for 2 years. Her body mass index was 14.2. After diagnosis, only abdominal distension and dyspepsia were clinically observed, without any significant relevant history. Physical examination suggested tenderness in the epigastrium, with no rebound tenderness. Excluding hypoproteinemia, no other abnormalities were clearly indicated after the laboratory examinations (assessment of tumor markers, routine blood, urine, and stool tests). Computed tomography [Figure 1] imaging illustrated about 8 cm × 8 cm × 4 cm cyst located in the lower esophagus compressing the stomach. An esophagus barium meal examination revealed that the cyst was exogenous [Figure 2].
Figure 1: Computed tomography demonstrated a huge cyst locates in the lower esophagus which compressed the stomach

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Figure 2: Esophagus barium meal examination revealed that the cyst is exogenous

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Surgical intervention revealed that the mass originated from the inferior esophagus and the stomach which was compressed by the cyst. A complete resection of the cyst was performed; a little pity is that we did not use a scale to remake the tumor [Figure 3]. Following surgical resection, the patient's abdominal distention and the dyspepsia ceased. The patient recovered well and was discharged from the hospital a week later. The pathology report documented a cystic wall containing esophageal squamous epithelium, along with one well-developed muscular layer. In addition, cartilage and bronchial mucus glands were identified, and the definitive diagnosis of esophageal bronchogenic cyst was confirmed [Figure 4]. Written informed consent was obtained from the patient for publication of this case report and any accompanying images.
Figure 3: The patient received complete resection of the cyst

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Figure 4: Hematoxylin and eosin staining of tissue demonstrated ciliated epithelium with an occasional cartilaginous component inside the cyst

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  Discussion Top


It is well known that the bronchogenic cyst is a congenital lesion, which typically originates at the primitive foregut with abnormal budding.[6] As reported in the literature, bronchogenic cysts occur in tissues of the mediastinum, lung parenchyma, pericardium, thymus, etc. However, they are rare to be found in the esophagus.[7] Esophageal duplication cysts are also rare congenital cystic masses resulting from an error in foregut budding in the developing embryo, with a reported incidence of 1 in 8200 autopsies.[8] Differentiation between bronchogenic cysts and esophageal duplication cysts is often nontrivial. Without pathological information, it is often difficult to make a clear diagnosis based only on preoperative testing. In general, 60% of cysts in the esophagus are located in the lower third, while the rest are located in the middle third and the upper third (17% and 23%, respectively).[9] In contrast to esophageal cysts, bronchogenic cysts are diagnosed based on the presence of cartilage. It is believed that Palmer's pathologic criteria may be useful for the differentiation of bronchogenic from esophageal duplication cysts: (1) an attachment to the esophageal wall, (2) the gastrointestinal tract epithelium exists, and (3) two layers of muscularis propria are presented.[10] In conclusion, it is meaningless to distinguish between bronchogenic cyst and esophageal duplication cyst without pathology. The standard therapy is surgical resection. Compared to the diagnosis of esophageal duplication cysts, the diagnosis of bronchogenic cysts must meet the following criteria: (1) the cyst is covered with one layer of muscularis propria, (2) the cyst has a ciliated epithelium, and (3) there should be an occasional cartilaginous component inside the cyst.[11] Sometimes, with computed tomography and magnetic resonance imaging, the type of cysts could be appropriately revealed. However, to distinguish cystic from solid masses, the means of endoscopic ultrasonography are considered as sensitive. It helps facilitate the diagnosis of intramural esophageal lesions, attributing to its ability to distinctly profile elements of the size, layer of origin, and the relationship of the cyst to the esophagus. Meanwhile, new reports have revealed that CA125 and CA199 are positive expressions in cyst sections as demonstrated by immunohistochemical staining. Therefore, the measurement of CA199 and CA125 levels may be of diagnostic value when considering a bronchogenic cyst of the esophagus.

Depending on the position of the cyst, different approaches are applied for the removal. The most common location of these cysts is in the middle and superior mediastinum.[9] Completely intra-abdominal esophageal bronchogenic and esophagogastric junction cysts are rare.[12] Thoracotomy or video-assisted thoracoscopic surgery is typically performed when the mass location is in the middle and superior mediastinum. Open or laparoscopic surgery is typically performed when the mass location is in inferior segmental esophagus and esophagogastric junction.[1],[13] As for submucosal tumors arising from the muscularis propria, the endoscopic submucosal tunnel dissection (ESTD) is a novel method.[14] Endoscopic ultrasound fine-needle aspiration (EUS-FNA) of the lesion can be performed, if a clear diagnosis is confirmed before the operation; however, antibiotic prophylaxis and proton-pump inhibitors should be considered to avoid infection and esophageal fistula.[15]

In conclusion, esophageal bronchogenic cysts and esophageal duplication cysts are rare. It is often difficult to make a clear diagnosis before surgery. We demonstrate this in the esophageal bronchogenic cyst case study we reported on, and preoperative diagnosis is sometimes inconsistent with pathological diagnosis. EUS is the best diagnostic tool capable of investigating duplication cysts since it can distinguish between solid and cystic lesions. EUS-FNA could be performed but the risk of EUS-FNA-related side effects should be considered, such as infection, esophageal fistula, and tumor implantation metastasis. There is also a risk for malignant transformation in adulthood. To prevent carcinogenesis in future, necessary resection is required and should be conducted as early as possible,[11],[16] yet, the optimal surgical method is patient specific.

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.
Altieri MS, Zheng R, Pryor AD, Heimann A, Ahn S, Telem DA. Esophageal bronchogenic cyst and review of the literature. Surg Endosc 2015;29 (10):3010-5.  Back to cited text no. 1
    
2.
Pasquer A, Djeudji F, Hervieu V, Rabeyrin M, Barth X. A rare retrorectal presentation of a bronchogenic cyst: A case report. Int J Surg Case Rep 2016;24:112-4.  Back to cited text no. 2
    
3.
Han CQ, Lin R, Yu J, Zhang Q, Zhang Y, Liu J, Ding Z, Hou XH. A case report of esophageal bronchogenic cyst and review of the literature with an emphasis on endoscopic ultrasonography appearance. Medicine (Baltimore) 2016;95 (11):e3111.  Back to cited text no. 3
    
4.
Kawashima S, Segawa O, Kimura S, Tsuchiya M, Henmi N, Hasegawa H, Fujibayashi M, Naritaka Y. A case of cervical esophageal duplication cyst in a newborn infant. Surg Case Rep 2016;2 (1):30.  Back to cited text no. 4
    
5.
Bagheri R, Asnaashari AM, Afghani R. Esophageal duplication cyst. Asian Cardiovasc Thorac Ann 2015;23 (3):332-4.  Back to cited text no. 5
    
6.
Aktoǧu S, Yuncu G, Halilçolar H, Ermete S, Buduneli T. Bronchogenic cysts: Clinicopathological presentation and treatment. Eur Respir J 1996;9 (10):2017-21.  Back to cited text no. 6
    
7.
Gou YW, Wang YT, Fang HY, Xu XM, Yu WY, Zhang KG, Yu Y. Bronchogenic cyst in the hepatogastric ligament masquerading as an esophageal mesenchymal tumor: A case report. Int J Clin Exp Pathol 2015;8 (11):15307-11.  Back to cited text no. 7
    
8.
Aldrink JH, Kenney BD. Laparoscopic excision of an esophageal duplication cyst. Surg Laparosc Endosc Percutan Tech 2011;21 (5):e280-3.  Back to cited text no. 8
    
9.
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 (12):854-6.  Back to cited text no. 9
    
10.
Martin ND, Kim JC, Verma SK, Rubin R, Mitchell DG, Bergin D, Yeo CJ. Intra-abdominal esophageal duplication cysts: A review. J Gastrointest Surg 2007;11 (6):773-7.  Back to cited text no. 10
    
11.
Fievet L, D'Journo XB, Guys JM, Thomas PA, De Lagausie P. Bronchogenic cyst: Best time for surgery? Ann Thorac Surg 2012;94 (5):1695-9.  Back to cited text no. 11
    
12.
Turkyilmaz A, Eroglu A, Subasi M, Findik G. Intramural esophageal bronchogenic cysts: A review of the literature. Dis Esophagus 2007;20 (6):461-5.  Back to cited text no. 12
    
13.
Tonouchi A, Kinoshita T, Sunagawa H, Hamakawa T, Kaito A, Shibasaki H, Kuwata T, Seki Y, Nishida T. Bronchogenic cyst at esophagogastric junction treated by laparoscopic full-thickness resection and hand-sewn closure: A case report. Surg Case Rep 2016;2 (1):41.  Back to cited text no. 13
    
14.
Tang XW, Jiang B, Gong W. Endoscopic submucosal tunnel dissection of a bronchogenic esophageal cyst. Endoscopy 2014;46(S 01):E626-7.  Back to cited text no. 14
    
15.
Somani P, Sharma M. Endoscopic ultrasound of esophageal duplication cyst. Indian J Gastroenterol 2016;35 (6):497-8.  Back to cited text no. 15
    
16.
Dai ZJ, Kang HF, Lin S, Bai MH, Ma L, Min WL, Lu WF, Wang XJ. Esophageal cancer with esophageal duplication cyst. Ann Thorac Surg 2013;96 (1):e15-6.  Back to cited text no. 16
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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