African Journal of Paediatric Surgery About APSON | PAPSA  
Home About us Editorial Board Current issue Search Archives Ahead Of Print Subscribe Instructions Submission Contact Login 
Users Online: 1647Print this page  Email this page Bookmark this page Small font size Default font size Increase font size 
 
 


 
CASE REPORT Table of Contents   
Year : 2010  |  Volume : 7  |  Issue : 3  |  Page : 203-205
Duplication of cervical oesophagus: A case report and review of literatures


1 Isfahan University of Medical Sciences, Al-Zahra Hospital, Isfahan, Iran
2 Isfahan University of Medical Sciences, Al-Zahra Hospital, Isfahan; Rafsanjan University of Medical Sciences, Rafsanjan, Kerman Province, Iran

Click here for correspondence address and email

Date of Web Publication18-Sep-2010
 

   Abstract 

Foregut duplication is commonly found in the posterior mediastinum. 10-20% of these anomalies are associated with oesophageal duplication. It can occur in all parts of oesophageal length. Although duplication of cervical oesophagus has been previously reported, but a majority of them were found in thoracic oesophagus. Infants with oesophageal duplication usually manifested by respiratory distress or asymptomatic thoracic mass, casually, detected in X-ray. A 7-month-old infant weighing about 7.5 kg, with the signboard of respiratory distress, fever and nutritional intolerance was admitted to our hospital. Physical examination showed dehydration, stridor, tachypnoea, intercostal retraction and neck stiffness. Plain chest radiogram showed dubious cystic mass in the distal neck. Cervical and chest computed tomography scan showed neck cystic lesion disseminated to posterior mediastinum, probably propagated to the respiratory system. Cystic lesion connected to oesophagus was partially resected, oesophagus was repaired and remaining mucous of a cyst was removed, then gastrostomy tube was applied.
Although cervical oesophageal duplication cysts are rare, but they must be considered as one of the differential diagnoses of cervical mass with respiratory distress in infants.

Keywords: Cyst, duplication of foregut, oesophageal duplication

How to cite this article:
Nazem M, Amouee A B, Eidy M, Khan IA, Javed H A. Duplication of cervical oesophagus: A case report and review of literatures. Afr J Paediatr Surg 2010;7:203-5

How to cite this URL:
Nazem M, Amouee A B, Eidy M, Khan IA, Javed H A. Duplication of cervical oesophagus: A case report and review of literatures. Afr J Paediatr Surg [serial online] 2010 [cited 2019 Nov 14];7:203-5. Available from: http://www.afrjpaedsurg.org/text.asp?2010/7/3/203/70429

   Introduction Top


Duplications can occur in all parts of the oesophageal length. Most of them are located in right thoracic and lower half segment of oesophagus. [1],[2] Cysts of oesophageal duplication are rare and at least 10 cases are reported till now. [3] Almost all these cases were detected in early days of life, manifested with respiratory distress and can be life threatening. If in case, clinical signs revealed before mass detection, tracheal intubation and urgent surgical interventions are needed. [3],[4]

Oesophageal duplication is divided into three types: cystic, tubular and diverticular form. Most of them are cystic. Tubular kinds in contrast with cystics belong to normal oesophagus. [1],[5] The importance of this case in addition to rarity is unusual sign and symptoms manifested by fever, neck stiffness and respiratory distress. These clinical features can be misleading and treated as meningitis.


   Case Report Top


A 7 month infant weighing 7.5 kg, presented with the respiratory distress, fever (38.5 C o or 101.3 o F), and nutritional intolerance. Physical examination revealed dehydration, feebleness, stridor, tachypnoea, intercostal retraction and neck stiffness. O 2 saturation was found to be 91% by pulse oximetry. She was previously hospitalized for 3 days due to seizure in another centre and treated by antibiotics and anticonvulsant without lumbar puncture.

At our hospital, patient's radiogram showed dubious cystic mass in the distal neck. Cervical and chest computed tomography scan showed cystic neck lesion disseminated to posterior mediastinum, probably propagated to the respiratory system. On physical exam no signs of swelling and tenderness were present in the neck.

Plain chest radiogram showed dubious cystic mass about 3 cm Χ 1 cm. Lateral cervical X-ray showed a cystic lesion with detached air-fluid level which caused anterior displacement of tracheal and oesophagus [Figure 1].
Figure 1 :Lateral cervical X-ray which shows the cystic lesion with air fluid level in the posterior of oesophagus and trachea.

Click here to view


According to cervical and chest computed tomography, lesion disseminated to posterior mediastinum, probably propagated to the respiratory system [Figure 2]. The duplication was resected and the oesophagus was repaired with interrupted absorbable sutures. A right thoracotomy was opened for mediastinal drainage and distal oesophageal duplication evaluation. A Stamm's gastrostomy was performed to allow the patient to eat. The excised oesophageal duplication was communicating with the oesophagus and consisted of a squamous cell epithelium and inflammatory cells infiltrated into the mucous membrane. The contents of the cyst were mucoid. Postoperatively, 3 months later the patient was doing well and able to eat a regular diet. The oesophagus-duplicated cyst was confirmed by histopathology.
Figure 2 :Cervical CT scan which shows the cystic lesion with air fluid level that seems connected to trachea.

Click here to view



   Discussion Top


Embryologically, duplications result from a defect in the tubulation (vacuolization) of the oesophagus, normally occurring in the sixth gestational week. [2],[3] As the foregut epithelium develops, it elongates, develops a lumen and undergoes dextro-rotations. Thus, the majority of oesophageal duplications occur distally and on the right. In the present case the oesophageal duplication was proximal to the cervical part and on the left.

The duplication of the oesophagus may be of three forms: (1) a cystic form [6] that may or may not communicate with the oesophageal lumen; (2) a tubular form [7],[8] or (3) a diverticular form. [9],[10] In one study, [11] only six tubular duplications of 44 oesophageal duplications were identified. In another review of alimentary tract duplications, [12] 22% were intrathoracic, and all were cystic rather than tubular.

The duplication of the hollow structures of the digestive tract manifest three essential points: (1) they are in intimate contact with the alimentary tract; (2) they are lined by a mucous membrane similar to other such membranes at the same level of the digestive tract and (3) they are provided with a smooth muscle coat, though an oesophageal duplication without muscle fibres has been reported. [13] In the present case, the wall between the cyst and oesophagus was complete with a smooth muscle layer.

Most tubular duplications were reported in late childhood or adults, comparatively asymptomatic and commonly connected to oesophagus, [5] but in our patient symptoms appeared during the infantile period.

It often appears in form of soft tissue mass in posterior mediastinum and exerts pressure on oesophagus. Chest and abdominal CT-scan or MRI is indicated, because some of mediastinal duplication maybe continuous under the diaphragm ultrasonography is recommended as a screening method for such cases. [2] Oesophagogram also helps in diagnosis. [2] Mentionable paraclinic tests used in our patient were: chest roentgenogram, lateral cervical X-Ray, cervical and chest CT-scan.

Oesophageal duplication may be present with some anomalies such as small intestinal duplication, oesophageal atresia distal to duplication site, tracheoesophageal fistula, scoliosis, hemivertebra, adhesion of spine and anterior spina bifida. [14],[15]

Because of caudal foregut and notochord cephalic development, spine defects basically are above the mediastinal cyst. [15] These are cysts found incidentally by chest roentgenogram in asymptomatic patients. [14] Finally, symptom appears in of cases when complications like infection, mass effects, bleeding, neoplasm and oesophageal or respiratory obstruction causing difficulties for patient. [14],[15] Infants affected by a cervical oesophageal duplication cyst, predominately had respiratory distress before detection of mass complications. These patients might confess malnutrition due to rapid growth of the cyst secondary to infection, bleeding and discharge to the cyst. [3]

Differential diagnoses for cervical cysts include lymphatic malformation, airway cyst, bronchial cyst or thyroglossal duct cyst sometimes present in the lateral of the neck. [3] Preferred treatment is complete resection of the cyst. In this way prognosis will be excellent. [3],[15] If complete excision is not possible, mucosa should be removed and subsequently the cyst gets dissolve. [3],[15]


   Conclusions Top


Although cervical oesophageal duplication cysts are rare, but they must be considered as one of the differential diagnoses of cervical mass with respiratory distress in infants.

 
   References Top

1.Kumar D, Samujh R, Rao KL. Infected esophageal duplication cyst simulating empyema. Indian Pediatr 2003;40:423-5.   Back to cited text no. 1  [PUBMED]  [FULLTEXT]  
2.Shew SB, Holcomb GW III. Alimentary tract duplications. In: Ashcraft KW, Murphy JP, Holcomb GW III, editors. Pediatric Surgery. 4 th ed. Amsterdam: Elsevier; 2005. p. 543-52.  Back to cited text no. 2      
3.Lund DP. Alimentary tract duplication. In: Grosfeld J, O'neill J, Fonkalsrud E, Coran A, editors. Pediatric surgery. Toronto: Judith Fletcher; 2006. p. 1389-98.  Back to cited text no. 3      
4.Wootton-Gorges SL, Eckel GM, Poulos ND, Kappler S, Milstein JM. Duplication of the cervical esophagus: A case report and review of the literature. Pediatr Radiol 2002;32:533-5.   Back to cited text no. 4  [PUBMED]  [FULLTEXT]  
5.Khan NZ, Mansoor C, Haq A, Qazi A. Tubular duplication of esophagus: Rare congenital malformations require individualized and innovative procedures. Pak J Mod Sci 2006;22:465-7.  Back to cited text no. 5      
6.Nakahara K, Fujii Y, Miyoshi S, Yoneda A, Miyata M, Kawashima Y. Acute symptoms due to a huge duplication cyst ruptured into the esophagus. Ann Thorac Surg 1990;50:309-11.  Back to cited text no. 6  [PUBMED]    
7.Cantallops JG, Adrover AO, Fernandez JM, Fernandez AB. Incomplete duplication of the esophagus: One case. Endoscopy 1981;13:46-8.  Back to cited text no. 7      
8.Dresler CM, Patterson GA, Taylor BR, Moote DJ. Complete foregut duplication. Ann Thorac Surg 1990;50:306-8.  Back to cited text no. 8  [PUBMED]    
9.Ohbatake M, Muraji T, Yamazato M, Higashimoto Y, Nishijima E, Tsugawa C. Congenital true diverticula of the esophagus: A case report. J Pediatr Surg 1997;32:1592-4.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]  
10.Gorenstein A, Serour F, Bujanover Y. Unusual presentation of esophageal communicating duplication in a child. J Pediatr Surg 1999;34:1430-1.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]  
11.Peiper M, Lambrecht W, Kluth D, Huneke B. Bleeding esophageal duplication detected in utero. Ann Thorac Surg 1995;60:1790-1.  Back to cited text no. 11      
12.Bajpai M, Mathur M. Duplications of the alimentary tract: Clues to the missing links. J Pediatr Surg 1994;29:1361-5.  Back to cited text no. 12  [PUBMED]  [FULLTEXT]  
13.Knight J, Garvin PJ, Lewis E Jr. Gastric duplication presenting as a double esophagus. J Pediatr Surg 1983;18:300-1.  Back to cited text no. 13  [PUBMED]  [FULLTEXT]  
14.Birmole BJ, Kulkarni BK, Vaidya AS, Borwankar SS. Intrathoracic enteric foregut duplication cyst. J Postgrad Med 1994;40:228-30.  Back to cited text no. 14  [PUBMED]  Medknow Journal  
15.Khan MU, Saidy KM, Ousulimane DM, Khan MD. Posterior mediastinal gastoenteric cyst in neonate. Saudi Med J 2004;25 :955-7 .  Back to cited text no. 15  [PUBMED]    

Top
Correspondence Address:
Ishfaq Abass Khan
Isfahan University of Medical Sciences, Isfahan
Iran
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0189-6725.70429

Rights and Permissions


    Figures

  [Figure 1], [Figure 2]

This article has been cited by
1 Esophageal Duplication Cyst Treated Thoracoscopically During the Neonatal Period
Barbara Cuch,Pawel Nachulewicz,Andrzej Pawel Wieczorek,Magdalena Wozniak,Elzbieta Pac-Kozuchowska
Medicine. 2015; 94(49): e2270
[Pubmed] | [DOI]
2 Anastomosis instead of resection: an unsual approach for the treatment of a cervical esophageal duplication cyst
Katharina Wenke,Konrad Reinshagen
Journal of Pediatric Surgery Case Reports. 2014;
[Pubmed] | [DOI]
3 Fehlbildungen des Ösophagus
C. Falkeis,T. Hager,K. Freund-Unsinn,J. Wohlschläger,L. Veits,J. Hager
Der Pathologe. 2013; 34(2): 94
[Pubmed] | [DOI]
4 A preoperative diagnosis of esophageal duplication cyst by endoscopic ultrasonography
Yamakawa, Y., Sato, H., Kusafuka, K., Kondo, H., Tsubosa, Y.
Japanese Journal of Gastroenterological Surgery. 2012; 45(12): 1153-1160
[Pubmed]
5 Foregut duplication cyst: An unusual presentation during childhood
Hammoud, A., Hourani, M., Akoum, M., Rajab, M.
North American Journal of Medical Sciences. 2012; 4(6): 287-289
[Pubmed]
6 Communicating esophageal tubular duplication in a newborn infant
Huang, Y., Wang, D., Liu, X., Wang, W., Zhang, Z.
Journal of Pediatric Surgery. 2011; 46(8): 1655-1657
[Pubmed]
7 Communicating esophageal tubular duplication in a newborn infant
Ying Huang,Dajia Wang,Xin Liu,Weiling Wang,Zhibo Zhang
Journal of Pediatric Surgery. 2011; 46(8): 1655
[Pubmed] | [DOI]



 

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


    Abstract
    Introduction
    Case Report
    Discussion
    Conclusions
    References
    Article Figures

 Article Access Statistics
    Viewed4671    
    Printed230    
    Emailed0    
    PDF Downloaded222    
    Comments [Add]    
    Cited by others 7    

Recommend this journal