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Journal of Indian Association of Pediatric Surgeons
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LETTER TO EDITOR
Year : 2017  |  Volume : 22  |  Issue : 3  |  Page : 193-195
 

Coexistence of both bronchogenic and esophageal duplication cyst in a single mass: An interesting coincidence


Department of Pathology, Lady Hardinge Medical College, New Delhi, India

Date of Web Publication8-Jun-2017

Correspondence Address:
Akanksha Jain
Resaerch Officer, All India Institute of Medical Science, New Delhi - 110 029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0971-9261.207628

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How to cite this article:
Jain A, Agarwal S, Kashyap A, Beniwal A. Coexistence of both bronchogenic and esophageal duplication cyst in a single mass: An interesting coincidence. J Indian Assoc Pediatr Surg 2017;22:193-5

How to cite this URL:
Jain A, Agarwal S, Kashyap A, Beniwal A. Coexistence of both bronchogenic and esophageal duplication cyst in a single mass: An interesting coincidence. J Indian Assoc Pediatr Surg [serial online] 2017 [cited 2017 Nov 18];22:193-5. Available from: http://www.jiaps.com/text.asp?2017/22/3/193/207628




Sir,

Bronchogenic and esophageal duplication cysts form as a result of defective budding, differentiation, or separation of the primitive foregut.[1] Till date, fewer than five cases of intramural esophagus bronchogenic cysts are reported.[2] The coexistence of esophageal duplication and bronchogenic cysts in a single mass is extremely rare. It represents anomalous development of both ventral and dorsal primitive foregut along with incomplete recanalization of esophagus. A 6-day-old male neonate presented to the pediatric emergency with progressive respiratory distress and recurrent vomiting. Computed tomography (CT) scan thorax showed a well-defined fluid-attenuated cystic lesion in posterior mediastinum, extending to left lower lung field [Figure 1]a. No extension to the vertebral column could be identified. During surgical excision, an extraluminal, intramural cyst was seen arising from the wall of lower esophagus not communicating with its lumen. On gross examination, cyst measured 4.2 cm × 3.5 cm × 2 cm was multiloculated, had smooth inner wall of variable thickness. Microscopically, cyst wall was lined by stratified squamous epithelial lining, had submucosal gastric glands and distinct double-layered smooth muscle wall, suggestive of esophageal duplication cyst [Figure 1]b. Adjacent areas of cyst wall showed pseudostratified ciliated columnar to flattened epithelium, with underneath seromucinous glands and cartilage suggestive of bronchogenic cyst [Figure 1]c. Both these cysts had common wall and were not communicating in each other [Figure 1]d. A diagnosis of bronchopulmonary foregut malformation with complete components of both bronchogenic and esophageal duplication cyst in a single mass was concluded.
Figure 1: Computed tomography scan of thorax showing a well-defined fluid-attenuated cystic lesion in posterior mediastinum (a), microphotograph showing esophageal duplication cyst lined by nonkeratinizing stratified squamous epithelium and double-walled smooth muscle wall (b), bronchogenic cyst lined by ciliated columnar epithelium and having cartilage and seromucinous glands in its wall (c), common wall between two cysts showing cartilage and gastric glands (d), (H and E, ×40)

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The term bronchopulmonary foregut malformation was coined by Gerle et al. in 1968.[2] It includes a wide spectrum of anomalies such as extra- and intra-lobar pulmonary sequestrations having partial or complete gastrointestinal communication, foregut diverticula, foregut duplications, tracheoesophageal fistulas, bronchoesophageal fistulas, and congenital cystic adenomatoid malformations of the lung.[3] In infants and children, foregut duplication cysts account for 15% of all mediastinal masses.[4] Two most common foregut cysts are bronchogenic and esophageal duplication cysts. In general, these are separate lesions but rarely can be seen in combinations.[1],[2],[3] Only a few case reports of coexisting lesions are described in the literature.[1],[3] Clinically foregut malformation usually present with respiratory distress, stridor, dyspnea, or dysphagia. CT scan is important diagnostic modality; however, histopathology is required for confirmation. In the index case, the presence of cartilage and distinct double-walled muscle layer points toward bronchogenic and esophageal duplication cyst, respectively. Ciliated columnar epithelial lining is described in both developing esophagus and trachea.[1],[2],[3],[4] Histopathological findings may lead to diagnostic dilemma of mature cystic teratoma; however, identification of esophageal wall origin and posterior mediastinal location at the time of surgery aided in the correct diagnosis. Foregut malformations are often complicated by infections (most common), ulceration, bleeding (in gastric mucosa lined foregut cysts), and rarely malignancy.[4] Surgical excision via open thoracotomy is the treatment of choice. However, for noncommunicating extraluminal bronchogenic and esophageal duplication cysts, laparoscopic enucleation is the preferred approach.[5] An accurate preoperative diagnosis is not possible by radiological investigations alone, and multidisciplinary approach is required for the confirmation.

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   References Top

1.
Horwitz JR, Lally KP. Bronchogenic and esophageal duplication cyst in a single mediastinal mass in a child. Pediatr Pathol Lab Med 1996;16:113-8.  Back to cited text no. 1
    
2.
Kitano Y, Iwanaka T, Tsuchida Y, Oka T. Esophageal duplication cyst associated with pulmonary cystic malformations. J Pediatr Surg 1995;30:1724-7.  Back to cited text no. 2
    
3.
Eom DW, Kang GH, Kim JW, Ryu DS. Unusual bronchopulmonary foregut malformation associated with pericardial defect: Bronchogenic cyst communicating with tubular esophageal duplication. J Korean Med Sci 2007;22:564-7.  Back to cited text no. 3
    
4.
Hasegawa S, Koga M, Matsubara T, Oga A, Furukawa S. Congenital cystic adenomatoid malformation complicated by esophageal duplication cyst in a 6-month-old girl. Pediatr Pulmonol 2002;34:398-401.  Back to cited text no. 4
    
5.
Ballouhey Q, Galinier P, Abbo O, Andrieu G, Baunin C, Sartor A, et al. The surgical management and outcome of congenital mediastinal malformations. Interact Cardiovasc Thorac Surg 2012;14:754-9.  Back to cited text no. 5
    


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