Remedy Publications LLC., | http://clinicsinsurgery.com/

Clinics in Surgery

2019 | Volume 4 | Article 26791

Intra-Abdominal Para-esophageal Bronchogenic Cyst: The Necessity for Surgical Intervention

OPEN ACCESS

*Correspondence:Edgar D Sy, Department of Pediatrics

Surgery, National Cheng Kung University Hospital, College of

Medicine, National Cheng Kung University, 138 Sheng-Li Rd, Tainan,

Taiwan, ROC,E-mail: edsyfalcon2@yahoo.com.tw

Received Date: 21 Nov 2019Accepted Date: 09 Dec 2019Published Date: 16 Dec 2019

Citation: Sy ED, Su P-J, Chu C-Y, Shan Y-S.

Intra-Abdominal Para-esophageal Bronchogenic Cyst: The Necessity for Surgical Intervention. Clin Surg. 2019;

4: 2679.

Copyright © 2019 Edgar D Sy. This is an open access article distributed

under the Creative Commons Attribution License, which permits unrestricted use, distribution, and

reproduction in any medium, provided the original work is properly cited.

Case ReportPublished: 16 Dec, 2019

AbstractBronchogenic Cyst (BC) is developmental abnormalities of the primitive foregut resulting from aberrant budding from the ventral diverticulum. As most intra-abdominal para-esophageal BC is clinically asymptomatic, it is usually diagnosed incidentally using radiological studies, either using sonogram or computed tomography. It is often difficult to differentiate from other intra-abdominal or retroperitoneal neoplastic lesions. Preoperative diagnosis for histological confirmation and absence of malignant degeneration may be done by image guided aspiration technique with symptomatic relief. However, surgical complete excision, rather than partial excision and aspiration, is the choice of treatment for prevention of recurrence. Long term follow-up with image study is necessary to detect recurrence at least for 2-3 decades.

Edgar D Sy1*, Ping-Jui Su2, Chang-Yao Chu3 and Yan–Shen Shan2

1Department of Pediatrics Surgery, National Cheng-Kung University Hospital, College of Medicine, National Cheng Kung University, Taiwan

2Department of Surgery, National Cheng-Kung University Hospital, College of Medicine, National Cheng Kung University, Taiwan

3Department of Pathology, National Cheng-Kung University Hospital, College of Medicine, National Cheng Kung University, Taiwan

IntroductionBronchogenic Cyst (BC) is developmental abnormalities of the primitive foregut resulting from

aberrant budding from the ventral diverticulum. As most intra-abdominal para-esophageal BC are clinically asymptomatic, is usually diagnosed incidentally. When diagnosed, either using sonogram or computed tomography, it is often difficult to differentiate from other intra-abdominal or retroperitoneal neoplastic lesions. Preoperative diagnosis may be done by image guided aspiration technique for histological confirmation of its benign nature and absence of malignant degeneration with symptomatic relief. However, surgical complete excision, rather than partial excision and aspiration, is the choice of treatment for prevention of recurrence.

Case PresentationThis is a 14-year-old male, who initially complained of intermittent post-prandial peri-umbilical

cramping pain with exacerbation of symptoms, especially when taking late night meal. However, except for anorexia, there were no other accompanied symptoms such as nausea, vomiting, diarrhea, constipation, fever, cough, dysuria, or body weight loss. Initial evaluation using abdominal sonogram showed a cystic mass, about 1.6 cm × 0.8 cm, over left lobe of liver, adjacent to the stomach cardia (Figure 1). Upper GI series, using barium, to evaluate the relationship of mass and stomach showed no sign of compression or filling defect of esophagus and stomach (Figure 2). Abdominal CT scan showed thin wall cystic lesion with 20-40 Hounsfield units (HU), size of 2.0 cm × 1.8 cm × 1.7 cm, located on the esophageo-cardial junction (Figure 3). Laboratory tests including complete blood counts, liver function test and blood chemistry were all within normal ranges. Tumor marker such as Carcino-Embryonic Antigen (CEA), Carbohydrate Antigen 19-9 (CA19-9), Cancer Antigen 72-4 (CA72-4), Alpha Fetoprotein (AFP) and Human Chorionic Gonadotropin (HCG) were not evaluated. The patient and his family decided for surgical excision for diagnostic confirmation. Intra-operative findings showed a cystic lesion, about 1.5 cm × 2 cm, located on the medial side of esophageo-cardial junction, containing non-acrid odor, light milky fluid (Figure 4). Histopathologic examination of the cyst showed that the wall of the cyst is lined with respiratory epithelial lining with underlying seromucinous glands with no cartilage tissue is found (Figure 5). Patient recovery uneventful from surgery and repeat examination using CT did not show evidence of recurrence at 6 month postoperatively.

Edgar D Sy, et al., Clinics in Surgery - Pediatric Surgery

Remedy Publications LLC., | http://clinicsinsurgery.com/ 2019 | Volume 4 | Article 26792

DiscussionBronchogenic cysts are congenital anomaly that arises from an

early error in the budding of the tracheal ventral diverticulum [1]. These are most commonly found in the mediastinum and may be arbitrarily divided into the following groups: 1. Paratracheal 2. Carinal 3. Hilar 4. Paraesophageal 5. Miscellaneous [2]. Numerous extra-mediastinal locations have been described, including cervical [3], cutaneous [4], intra-abdominal [5-9], distal esophagus [9], and retroperitoneal locations [10] In human beings, the thoracic and abdominal cavities are linked via the pericardio-peritoneal canal during early embryonic life and between 26 and 40 days of embryogenesis is divided by the fusion of the pleuroperitoneal membranes, in which a portion of the tracheobronchial tree may be pinched off and migrate, resulting in an exceptionally unusual intra-abdominal BC [7]. BC is predominantly

found on left of the midline, either retroperitoneal or intra-peritoneal location, such as crus of diaphragm [8], distal esophagus [9], stomach [5], omental bursa [11], gall bladder surface [6], distal pancreas [7], and adrenal gland [12]. Diagnosis is seldom done preoperatively, despite being suspect by studies in about 50% [1]. Often, these lesions are discovered incidentally due to absence of symptoms or during abdominal sonographic evaluation of abdominal discomfort, pain or sign of gastrointestinal obstruction, wherein that of size cyst is enough to compress the adjacent viscera. In thoracic BC, the size tend to be larger in symptomatic than asymptomatic cases. The content of BC, such as the density, amount of proteinaceous substances and amount of calcium, usually affect the finding of radiologic studies [13,14]. The location of a cyst on the left of midline on imaging studies such as sonogram, computed tomography or magnetic resonance imaging should suspect its presence [15] and the shape of the mass usually varied due to its cyst nature and may comfort with the anatomic space availability. As in this case, the cyst mass imprint of the surface of the liver which lead to suspicious of liver cyst. Sonogram may either showed hypo-echoic [16], anechoic [17,18] lesion and presence of free calcium in the cystic mass may help in diagnosis [19]. The characteristic finding on computed tomography scan is a hyper-dense homogeneous mass, with Hounsfield score ranging from 0 to 90 unit [13,14,20] while magnetic resonance imaging showed a homogeneous mass either with high-to-decreased signal on T1-weighted images and intermediate-increased signal on T2-weighted images [21,22]. Histological characteristics of typical BC may include the following: Cyst wall may be thickened, with hyalinized basement membrane, elastic fibers, fibrous connective tissue, smooth muscle and cartilage. The epithelial lining is composed of pseudostratified, cuboidal or columnar ciliated epithelium, with mucus-secreting

Figure 1: Abdominal sonogram showed a cystic mass, about 1.6 cm × 0.8 cm, over left lobe of liver, adjacent to the stomach cardia.

Figure 2: Upper GI series, using barium showed no sign of compression or filling defect of esophagus and stomach.

Figure 3: Abdominal CT scans in upper axial and lower coronal view showed thin wall cystic lesion, about 2.0 cm × 1.8 cm × 1.7 cm, located on the esophageo-cardial junction without evidence of intraluminal continuity.

Figure 4: Intra-operative findings showed a cystic lesion, about 1.5 cm × 2 cm, located on the medial side of esophageo-cardial junction.

Figure 5: Histopathologic examination showed that the wall of the cyst is lined with respiratory epithelial lining (arrow head) with underlying seromucinous glands (arrow).

Edgar D Sy, et al., Clinics in Surgery - Pediatric Surgery

Remedy Publications LLC., | http://clinicsinsurgery.com/ 2019 | Volume 4 | Article 26793

glands. Immunostaining study such as respiratory mucin may direct support the diagnosis while CEA and CA199 may be elevated in some cases, however, it was not done in this patient [23,24].

Preoperative pathologic diagnosis is possible by radiologic fine needle aspiration, is a reliable, sensitive and specific method for diagnostic evaluation with high accuracy of about 80 percent [25] and can also use symptomatic relief of symptoms, however, the risk of seeding, rupture, infection and late recurrence may complicates future surgery [26-29]. Surgical total excision is curative and preferred method of management compared to partial excision or aspiration, in order to established a definitive histological diagnosis of its benign and exclude the presence of malignancy [3,30,31]. Incomplete resection leads to disease recurrence [29] and a possibility that may progress to malignant lesions. Either conventional open or laparoscopic approach can be used would depends on the surgeon experienced. The later has a number of advantages, such as less invasiveness and better postoperative recovery as well as a shorter hospital stay. Long term follow BC is necessary by imaging study, for either asymptomatic or symptomatic, that is drain, partially or completely excised, to detect recurrence and malignancy [23,29,30] for at least 2-3 decades [3,32].

ConclusionIntra-abdominal BC is usually diagnosed incidentally and rarely

symptomatic except when large enough to cause compression or obstruction of adjacent anatomic structure. A complete surgical excision for histologic diagnosis of its benign nature is preferred method of treatment to avoid recurrence rather than partial excision or aspiration. Long term follow up with image study is necessary to detect recurrence at least for 2-3 decades.

References1. St-Georges R, Deslauriers J, Duranceau A, Vaillancourt R, Deschamps

C, Beauchamp G, et al. Clinical spectrum of bronchogenic cysts of the mediastinum and lung in the adult. Ann Thorac Surg. 1991;52(1):6-13.

2. Maier HC. Bronchiogenic Cysts of the Mediastinum. Ann Surg. 1948;127(3):476-502.

3. Ramenofsky ML, Leape LL, McCauley RG. Bronchogenic cyst. J Pediatr Surg. 1979;14(3):219-24.

4. Kim NR, Kim HH, Suh YL. Cutaneous bronchogenic cyst of the abdominal wall. Pathol Int. 2001;51(12):970-3.

5. Sato M, Irisawa A, Bhutani MS, Schnadig V, Takagi T, Shibukawa G, et al. Gastric bronchogenic cyst diagnosed by endosonographically guided fine needle aspiration biopsy. J Clin Ultrasound. 2008;36(4):237-9.

6. Kim KH, Kim JI, Ahn CH, Kim JS, Ku YM, Shin OR, et al. The first case of intraperitoneal bronchogenic cyst in Korea mimicking a gallbladder tumor. J Korean Med Sci. 2004;19(3):470-3.

7. Sumiyoshi K, Shimizu S, Enjoji M, Iwashita A, Kawakami K. Bronchogenic cyst in the abdomen. Virchows Arch A Pathol Anat Histopathol. 1985;408(1):93-8.

8. Mubang R, Brady JJ, Mao M, Burfeind W, Puc M. Intradiaphragmatic Bronchogenic Cysts: Case Report and Systematic Review. J Cardiothorac Surg. 2016;11(1):79.

9. Cheng Y, Chen D, Shi L, Yang W, Sang Y, Duan S, et al. Surgical treatment of an esophageal bronchogenic cyst with massive upper digestive tract hematoma without esophagectomy: a case report and the review of the literature. Ther Clin Risk Manag. 2018;14:699-707.

10. Byers JT, Gertz HE, French SW, Wang L. Case report: Retroperitoneal

bronchogenic cyst as a diagnostic dilemma after colon cancer diagnosis. Exp Mol Pathol. 2018;104(2):158-60.

11. Fan RY, Li N, Yang GZ, Sheng JQ. Bronchogenic cyst in the omental bursa: A case report. J Dig Dis. 2016;17(1):52-4.

12. Swanson SJ 3rd, Skoog SJ, Garcia V, Wahl RC. Pseudoadrenal mass: unusual presentation of bronchogenic cyst. J Pediatr Surg. 1991;26(12):1401-3.

13. Fischbach R, Benz-Bohm G, Berthold F, Eidt S, Schmidt R. Infradiaphragmatic bronchogenic cyst with high CT numbers in a boy with primitive neuroectodermal tumor. Pediatr Radio. 1994;24(7):504-5.

14. Nakata H, Nakayama C, Kimoto T, Nakayama T, Tsukamoto Y, Nobe T, et al. Computed tomography of mediastinal bronchogenic cysts. J Comput Assist Tomogr. 1982;6(4):733-8.

15. Haddon MJ, Bowen A. Bronchopulmonary and neurenteric forms of foregut anomalies. Imaging for diagnosis and management. Radiol Clin North Am. 1991;29(2):241-54.

16. Lee HS, Park CK, Joo KB, Shin HJ, Kim Y, Park DW, et al. Subcutaneous bronchogenic cyst: unusual unltrasonographic findings. J Ultrasound Med. 2001;20(5):563-6.

17. De Catte L, De Backer T, Delhove O, Mares C. Ectopic bronchogenic cyst: sonographic findings and differential diagnosis. J Ultrasound Med. 1995;14(4):321-3.

18. Rahmani MR, Filler RM, Shuckett B. Bronchogenic cyst occurring in the antenatal period. J Ultrasound Med. 1995;14(12):971-3.

19. Cornell SH. Calcium in the fluid of mediastinal bronchogenic cyst: a new roentgenographic finding. Radiology. 1965;85(5):825-8.

20. Chehade LK, Lambert V, Kollias J, Otto S. Bronchogenic cyst: the rarest adrenal incidentaloma. ANZ J Surg. 2018;88(3):243-5.

21. El Youssef R, Fleseriu M, Sheppard BC. Adrenal and pancreatic presentation of subdiaphragmatic retroperitoneal bronchogenic cysts. Arch Surg. 2010;145(3):302-4.

22. Castro R, Oliveira MI, Fernandes T, Madureira AJ. Retroperitoneal bronchogenic cyst: MRI findings. Case Rep Radiol. 2013;2013:853795.

23. Cuypers P, De Leyn P, Cappelle L, Verougstraete L, Demedts M, Deneffe G. Bronchogenic cysts: a review of 20 cases. Eur J Cardiothorac Surg. 1996;10(6):393-6.

24. Itoh H, Shitamura T, Kataoka H, Ide H, Akiyama Y, Hamasuna R, et al. Retroperitoneal bronchogenic cyst: report of a case and literature review. Pathol Int. 1999;49(2):152-5.

25. Assaad MW, Pantanowitz L, Otis CN. Diagnostic accuracy of image-guided percutaneous fine needle aspiration biopsy of the mediastinum. Diagn Cytopathol. 2007;35(11):705-9.

26. Lundstedt C, Stridbeck H, Andersson R, Tranberg KG, Andren-Sandberg A. Tumor seeding occurring after fine-needle biopsy of abdominal malignancies. Acta Radiol. 1991;32(6):518-20.

27. Tyagi R, Dey P. Needle tract seeding: an avoidable complication. Diagn Cytopathol. 2014;42(7):636-40.

28. Onuki T, Kuramochi M, Inagaki M. Mediastinitis of bronchogenic cyst caused by endobronchial ultrasound-guided transbronchial needle aspiration. Respirol Case Rep. 2014;2(2):73-5.

29. Miller DC, Walter JP, Guthaner DF, Mark JB. Recurrent mediastinal bronchogenic cyst. Cause of bronchial obstruction and compression of superior vena cava and pulmonary artery. Chest. 1978;74(2):218-20.

30. Sullivan SM, Okada S, Kudo M, Ebihara Y. A retroperitoneal bronchogenic cyst with malignant change. Pathol Int. 1999;49(4):338-41.

31. Brassesco MS, Valera ET, Lira RC, Torres LA, Scrideli CA, Elias J, et al. Mucoepidermoid carcinoma of the lung arising at the primary site of a

Edgar D Sy, et al., Clinics in Surgery - Pediatric Surgery

Remedy Publications LLC., | http://clinicsinsurgery.com/ 2019 | Volume 4 | Article 26794

bronchogenic cyst: clinical, cytogenetic, and molecular findings. Pediatr Blood Cancer. 2011;56(2):311-3.

32. Read CA, Moront M, Carangelo R, Holt RW, Richardson M. Recurrent

bronchogenic cyst. An argument for complete surgical excision. Arch Surg. 1991;126(10):1306-8.