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  Table of Contents    
CASE REPORT  
Year : 2013  |  Volume : 56  |  Issue : 4  |  Page : 411-412
Perianal bronchogenic cyst: A report of two cases


1 Department of General Surgery; Depatment of Colorectal Surgery, Beijing Friendship Hospital, Capital Medical University, Beijing, China
2 Depatment of Colorectal Surgery, Beijing Erlonglu Hospital, Beijing, China
3 Department of General Surgery, Beijing Friendship Hospital, Capital Medical University, Beijing, China
4 Department of Pathology, Beijing Erlonglu Hospital, Beijing, China

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Date of Web Publication18-Jan-2014
 

   Abstract 

Cutaneous presentations of bronchogenic cysts are rare in all age groups, particularly in adults. Previous reports showed that cutaneous manifestations of bronchogenic cysts were frequently presented above abdominal region. Here we present two cases of a perianal bronchogenic cyst. To the best to our knowledge, this is the first report of a bronchogenic cyst in this area.

Keywords: Bronchogenic cysts, cutaneous, perianal

How to cite this article:
Liu L, Zhang Y, Zhang Z, Si L. Perianal bronchogenic cyst: A report of two cases. Indian J Pathol Microbiol 2013;56:411-2

How to cite this URL:
Liu L, Zhang Y, Zhang Z, Si L. Perianal bronchogenic cyst: A report of two cases. Indian J Pathol Microbiol [serial online] 2013 [cited 2019 Dec 11];56:411-2. Available from: http://www.ijpmonline.org/text.asp?2013/56/4/411/125355



   Introduction Top


Bronchogenic cysts (BCs) are benign cystic lesions that originate from an accessory lung bud of the primitive foregut. Cutaneous presentations of BCs are rare in all age groups, particularly in adults. Previous reports of cutaneous lesions have been described as nodular and adherent and most frequently have a supraumbilical location. Here, we report two male adults with unique presentation of BC located on the perianal skin.


   Case Report Top


Case 1

A 53-year-old Asian man presented with a 5-year history of an asymptomatic perianal mass. Physical examination revealed a 3.5 by 4.0 by 4.0 cm bulbous spherical mass which was connected by a 2 cm stalk to the right anal edge. The mass was smooth, non-tender, and freely mobile. The overlying skin was hyperpigmented [Figure 1]a].

The lesion was completely removed by elliptical excision under local anesthesia. Postoperative complications were absent. The cut surface was found to contain a 3.5 cm diameter cyst with a smooth lining filled with clear and viscous fluid. Microscopic examination of the surgical specimen showed smooth muscle and mucous glands within the cyst wall [Figure 1]b. H&E staining revealed benign pseudostratified columnar epithelium without definite cilia identified [Figure 1]c. Immunochemistry of the cyst epithelial lining was strongly positive for cytokeratin 7 (CK7) [Figure 1]d. However, it was negative for thyroid transcription factor-1 (TTF-1), cytokeratin 20 (CK20), and CDX2.
Figure 1: (a) The mass was smooth, non-tender, and freely mobile. (b) Smooth muscle and mucous glands are present within the cyst wall (H & E, ×200). (c) Benign pseudostrati fi ed, columnar epithelium (H & E, ×400). (d) The cyst epithelial lining is positi ve for CK7 (×200). (e) Bulbous spherical mass connected by a 1 cm stalk to the right anal edge. (f) The cut surface was found to contain a 3 cm diameter cyst with a smooth lining fi lled with clear, viscous fl uid and brown necrotic tissue. (g) Benign columnar epithelium with cilia. (h) The cyst epithelial lining was strongly positi ve for CK7 (×200). (i) Rare cells at the cyst epithelial lining showed positi ve staining for TTF-1 (×400).

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Case 2

A 59-year-old Asian man was referred for an asymptomatic mass on his right perianal skin, which was present for 40 years. His lesion is reported to grow slowly and painless, while affects his movement. Physical examination revealed a 3.0 by 4.0 by 4.5 cm bulbous spherical mass connected by a 1 cm stalk to the right anal edge. It was smooth, non-tender, and mobile. The overlying skin was hyperpigmented without evidence of active drainage, ulceration, or infection [Figure 1]e.

The lesion was completely removed by elliptical excision under local anesthesia. There was no epithelial tract or mucoid-containing substance within the stalk of the lesion. Postoperative complications were absent. Gross examination of the surgical specimen showed a pedunculate skin fragment. The cut surface was found to contain a 3 cm diameter cyst with a smooth lining filled with clear, viscous fluid and brown necrotic tissue [Figure 1]f. Microscopic examination of the surgical specimen showed smooth muscle within the cyst wall. H&E staining revealed benign columnar epithelium with cilia [Figure 1]g. Immunochemistry of the cyst epithelial lining was strongly positive for CK7 [Figure 1]h. Rare cells were stained positively for TTF-1 [Figure 1]i. Both CK20 and CDX2 staining were negative throughout the section.


   Discussion Top


Given our limited knowledge for BC development and natural history, the disease is generally believed to result from abnormal budding of the ventral foregut from the tracheobronchial tree during the first 16 weeks of development. [1] More specifically, the cyst may either separate from the developing tracheal bud during sternal closure, or the cyst may separate from the parent bronchus before closure of the sternum and then migrate to a cutaneous region along the midline of the body surface. [2] Another mechanism may be the in situ development of the respiratory epithelium because of metaplasia of mature preexisting cutaneous tissue and primary anomalous differentiation (heterotopia) in the developing skin. [3]

BCs are benign cystic lesions. Although a few cases of malignancies have been reported, they are commonly found along the tracheobronchial tree. The most common locations for BCs are lung and the mediastinum in the carina. The locations for BCs in descending order of frequency are cutaneous, retroperitoneal, cervical, intradiaphragmatic, intrapericardial, intraspinal, intracardiac and intrapleural cavity. [4] Shah et al.[5] summarized a report of 86 cases of cutaneous or subcutaneous cysts in the literature. In most cases, the lesions are noted either shortly after birth or in early childhood. Most of them were located in the suprasternal notch (30%), and followed by the anterior neck, the scapular, and presternal areas. A cutaneous BC located in the perianal area has not been described in the English literature.

Remarkably, all the cutaneous BCs are located subcutaneously. The diagnosis of BC is based on examination of routine H&E histologic sections. Smooth muscle and sometimes cartilage are identified within the cyst wall. The lining exhibits tracheobronchial differentiation with respiratory-type epithelium, which includes columnar, ciliated, and mucus-secreting cells. In cases where the differential remains broad, immunohistochemical analysis may provide valuable input. In most reports, all BCs express CK7, and none expressed CK20 [6],[7],[8],[9] as well as CDX-2. [7],[8] In rare cases which expressed TTF-1, [10] the positive immunohistochemical staining for CK7 and negative staining for CK20 help narrow the differential diagnosis and support the pulmonary origin of these tumors. Combination of CK7, CK20, CDX2 and TTF-1 is a reliable immunohistochemical panel for BCs. [7],[8]

Although there is some controversy regarding surgical excision of BC, particularly when they are asymptomatic, it is generally believed that most of the cysts will ultimately become symptomatic with either mass effect, drainage, infection, or evolve into complex cysts. [1],[2],[3],[4],[6] Malignant degeneration of the cyst, including the development of melanoma, has been reported, [2],[3] which further supports the decision for early excision. [2],[4],[6]

 
   References Top

1.Correia-Pinto J, Gonzaga S, Huang Y, Rottier R. Congenital lung lesions: Underlying molecular mechanisms. Semin Pediatr Surg 2010;19:171-9.   Back to cited text no. 1
    
2.Carden C, Dykes E, Pollock AM. An unusual midline swelling: Case report of cutaneous bronchogenic cyst. Eur J Pediatr Surg 2008;18:345-6.  Back to cited text no. 2
    
3.Van der Putte SC, Toonstra J. Cutaneous 'bronchogenic' cyst. J Cutan Pathol 1985;12:404-9.  Back to cited text no. 3
    
4.Lee T, Tsai IC, Tsai WL, Jan YJ, Lee CH. Bronchogenic cyst in the left atrium combined with persistent left superior vena cava: The first case in the literature. AJR Am J Roentgenol 2005;185:116-9.  Back to cited text no. 4
    
5.Shah SK, Stayer SE, Hicks MJ, Brandt ML. Suprasternal bronchogenic cyst. J Pediatr Surg 2008;43:2115-7.  Back to cited text no. 5
    
6.Chung JH, Lim GY, Lee YS. A congenial cutaneous bronchogenic sinus of the abdominal wall in an infant. Pediatr Surg Int 2009;25:105-8.  Back to cited text no. 6
    
7.Roma A, Varsegi M, Magi-Galluzzi C, Ulbright T, Zhou M. The distinction of bronchogenic cyst from metastatic testicular teratoma: A light microscopic and immunohistochemical study. Am J Clin Pathol 2008;130:265-73.   Back to cited text no. 7
    
8.Kim PS, Cataletto M, Garnet DJ, Alexeeva V, Selbs E, Katz DS, et al. Unusual presentation of a cutaneous bronchogenic cyst in an asymptomatic neonate. J Pediatr Surg 2012;47:E9-12.  Back to cited text no. 8
    
9.Sagar J, Sagar B, Patel AF, Shak DK. Ciliated median raphe cyst of perineum presenting as perianal polyp: A case report with immunohistochemical study, review of literature, and pathogenesis. ScientificWorldJournal 2006;6:2339-44.  Back to cited text no. 9
    
10.Govaerts K, Van Eyken P, Verswijvel G, Van der Speeten K. A bronchogenic cyst, presenting as a retroperitoneal cystic mass. Rare tumors 2012;4:e13.  Back to cited text no. 10
    

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Correspondence Address:
Zhongtao Zhang
Department of General Surgery, Beijing Friendship Hospital, Capital Medical University, 95 Yongan Road, Xicheng District, Beijing - 100 050
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.125355

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