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 ¤ Introduction
 ¤ Case Report
 ¤ Discussion
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UNUSUAL CASE
Year : 2022  |  Volume : 18  |  Issue : 3  |  Page : 472-474
 

Successful thoracoscopic resection of a giant mediastinal cyst in a newborn boy


Department of Neonatal Surgery, Xi'an Children Hospital, Xi'an City, Shaanxi Province, China

Date of Submission29-Jul-2021
Date of Acceptance10-Nov-2021
Date of Web Publication11-Feb-2022

Correspondence Address:
Chaoxiang Lu
Department of Neonatal Surgery, Xi'an Children Hospital, 69 Xiyuyuanxiang, Xi'an 710003, Shaanxi Province
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmas.jmas_251_21

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 ¤ Abstract 


There are fewer reports of thoracoscopic surgical resection of mediastinal cysts in neonates. The aim of this article is to report on the feasibility of thoracoscopic resection of a large mediastinal cyst and the management of chylothorax after surgery in neonates.


Keywords: Chylothorax, mediastinal cyst, therapy, thoracoscopy


How to cite this article:
Li Z, Xie W, Lu C, Wang Q. Successful thoracoscopic resection of a giant mediastinal cyst in a newborn boy. J Min Access Surg 2022;18:472-4

How to cite this URL:
Li Z, Xie W, Lu C, Wang Q. Successful thoracoscopic resection of a giant mediastinal cyst in a newborn boy. J Min Access Surg [serial online] 2022 [cited 2022 Sep 28];18:472-4. Available from: https://www.journalofmas.com/text.asp?2022/18/3/472/337609





 ¤ Introduction Top


Over the past decade, thoracoscopy has become widely accepted as the method for the treatment of thoracic and mediastinal disorders for increasingly complex diagnoses and treatments.[1] However, even though there is a large body of literatures in the adult on thoracoscopic resection of large mediastinal masses via a thoracoscopic approach[2],[3] there are fewer reports of thoracoscopic surgical resection of mediastinal tumours in neonates. The aim of this article is to report on the feasibility of thoracoscopic resection of a large mediastinal cyst and the management of chylothorax after surgery in neonates.


 ¤ Case Report Top


A newborn boy was admitted to the hospital on the 8th day after birth with increased respiration, followed by respiratory distress and hypoxaemia with paroxysmal cyanosis, with hypoxia ameliorated by lying on the affected side. This full-term infant was not found to be abnormal before delivery. On admission, chest radiographs showed a homogeneous mass occupying the entire chest [Figure 1]. Computed tomography showed a cystic mass with irregular enhancement involving the entire right thorax, and magnetic resonance imaging suggested a giant cystic mass deformity. Laboratory tests for methaemoglobin and neuroenolase were normal. This baby was admitted to the operating room at 24 days old. Thoracoscopic resection was performed using a prone position with the right side elevated, a telescope trocar was placed in the fifth intercostal space in the subscapular angle line and two operating trocars were placed in the fourth and seventh intercostal spaces in the anterior axillary line. The pneumothorax pressure was set to 4 mmHg and the flow rate was set to 3 L/min. The cyst was first incised in a small section with electric knife and the fluid was drawn out from the cyst. The cyst was diminished and subsequently removed through the enlarged trocar hole after complete resection of the cyst along the margins. The operation lasted 147 min.
Figure 1: (a and b) Magnetic resonance imaging revealed a large cystic mass in the mediastinum (c) Upper boundary of tumour (d) Lower boundary of tumour (e) Post-operative appearance (f) The figure of the cyst

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Postoperatively, the child received ventilator support for 120 h with pressure-controlled mechanical ventilation. On the 3rd day after operation, an increased pleural effusion began to appear, which gradually worsened. On the 9th day after operation, the pleural effusion gradually increased to 270 ml, while the pleural effusion became milky white with positive Sudanese trichrome staining. On the 11th day after operation, the pleural drain was blocked and a replacement pleural drain was given. On the 13th day after the operation, the first intrathoracic injection of a sclerotherapy consisting of erythromycin and lidocaine plus 50% glucose was given. After the injection, the chest drain was clamped and the position was changed continuously. Since the pleural effusion persisted, two injections were given on day 16th and 19th day postoperatively. The pleural effusion disappeared on the 22nd post-operative day [Figure 2] and was subsequently fed with medium-chain fatty acid formula and discharged on the 26th post-operative day. One month later, he was fed with normal milk, but no pleural effusion appeared. Histopathological examination shows the presence of smooth muscle and enteric mucosa in the walls of the lesion, which suggests enterogenous origin [Figure 3]. Within 3 months of follow-up, the cyst recurred and chylothorax did not reappear.
Figure 2: (a) The thoracic drainage tube is blocked and the drainage fluid drops and the drainage tube is replaced (b) First time of intrapleural injection of sclerotherapy (c) Second time of intrapleural injection of sclerotherapy (d) Third time of intrapleural injection of sclerotherapy

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Figure 3: Pathology suggested intestinal mucosa is attached to the smooth muscle

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 ¤ Discussion Top


Mediastinal enterogenous cyst, also been called a duplication cyst, is a rare congenital malformation. It can be present with severe cardiorespiratory compromise including respiratory distress, shock, cardiac failure or arrhythmia in the neonates, which may require immediate surgical intervention soon after birth.[4] Moderate-to-severe acute respiratory distress occurs in approximate 67% of the cases, whereas all the patients have mass present on chest radiograph. About 15% of patients are asymptomatic and 40% present symptomatic by 1 month of age.

Enterogenous cysts are foregut duplication cysts occasionally associated with a vertebral defects.[5] Vertebral defects, mainly fissures or fusions, occur in about 21% of the patients.[5] These cysts may be located in prevertebral, vertebral or postvertebral positions. In this case, a vertebral defect is presented in prevertebral. In most cases, enterogenous cysts are seen in the mediastinum, peritoneal cavity, spinal canal, subarachnoid space and cerebral ventricle, but can also be seen in testis rarely.

About 36% enterogenous cysts are foregut in origin, 50% are midgut, 12% are hindgut and 2% are retroperitoneal, spinal, oropharyngeal or biliary in origin.[5] Enterogenous cysts include oropharyngeal, oesophageal, thoracoabdominal, gastric, duodenal and retroperitoneal. Oesophageal duplication represents abnormal budding of the dorsal primitive foregut and is the second most common type of foregut duplication cysts. Neurenteric cysts are oesophageal duplication cysts that extend into the spinal canal.

Histological examination of this cyst shows the presence of smooth muscle and enteric mucosa in the walls of the lesion. The mucosal type may differ from that of the gut to which it lies adjacent.

The differential diagnoses of mediastinal enterogenous in infancy include rhabdomyoma, teratoma, fibroma, bronchogenic cyst and rarely hemangioma, lipoma, thymic cyst, extralobar pulmonary sequestration, ectopic mediastinal pancreas and intrapericardial cysts.[6],[7]

Few papers report cases of successful resection by thoracoscopy in the neonatal group. Giant mediastinal cysts of intestinal origin are not common in children with large tumours and make surgical resection difficult. Since the cysts come from the intestinal origin, it can be shrunk by aspiration of the cystic fluid. The thoracoscope can be reoriented to better expose the tumour at the mediastinal junction and to resect cysts. The operation should be started by the upper edge of the tumour. After the upper edge of the cyst has been incised, the cyst can be lifted to expose the root of the cyst and complete excision is not difficult.

Although thoracic surgery may lead to a certain incidence of chylothorax,[8],[9] we have to avoid this complication. If chylothorax occurs, it can be treated by prohibiting diet or switched to medium-chain fatty acid-rich formula feeding to reduce fluid. The mixture of erythromycin with hypertonic glucose into the thorax can be used to accelerate pleural adhesion to cure chylothorax. The addition of lidocaine can reduce the pain of pleural reactions, and multiple injections can be given if once intrathoracic injection does not work.


 ¤ Conclusion Top


Although this is an extreme case presentation of mediastinal cyst, we suggest that thoracoscopic surgery is safe for neonates. Intrathoracic curing agent injection can be used to manage post-operative chylothorax once it occurs.

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.
Makdisi G, Roden AC, Shen KR. Successful resection of giant mediastinal lipofibroadenoma of the thymus by video-assisted thoracoscopic surgery. Ann Thorac Surg 2015;100:698-700.  Back to cited text no. 1
    
2.
Partrick DA, Rothenberg SS. Thoracoscopic resection of mediastinal masses in infants and children: An evaluation of technique and results. J Pediatr Surg 2001;36:1165-7.  Back to cited text no. 2
    
3.
Tan TW, Kim DS, Wallach MT, Mangray S, Luks FI. Thoracoscopic resection of a giant thymolipoma in a 4-year-old girl. J Laparoendosc Adv Surg Tech A 2008;18:903-5.  Back to cited text no. 3
    
4.
Modak A, Dhir SK, Khan AA, Rastogi P, Peters NJ, Mukhopadhyay K. Antenatally detected multiple mediastinal enterogenous cysts in a newborn presenting with severe cardio-respiratory compromise and early life-saving management. J Indian Assoc Pediatr Surg 2021;26:192-4.  Back to cited text no. 4
  [Full text]  
5.
Jain N, Gupta V, Mathur NB, Kumar A, Khurana N, Sarin YK. Enterogenous cyst presenting as mediastinal mass in a newborn. J Indian Assoc Pediatr Surg 2019;24:72-4.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Collison SP, Tomar M, Shrivastava S, Iyer KS. A rare intrapericardial enterogenous cyst presenting in infancy. Ann Thorac Surg 2006;81:e11-2.  Back to cited text no. 6
    
7.
Chojnacka H, Giżewska-Kacprzak K, Grodzki T, Rybkiewicz M, Nowakowski P, Gawrych E. Rare localization of an extralobar pulmonary sequestration in a child as a diagnostic challenge: A case report and review of the literature. Turk J Pediatr 2014;56:203-7.  Back to cited text no. 7
    
8.
Zheng J, Chen YY, Zhang CY, Zhang WQ, Rao ZY. Incidence and nutritional management of chylothorax after surgery for congenital heart diseases in children. Heart Surg Forum 2020;23:E902-6.  Back to cited text no. 8
    
9.
Zhang M, Wu W, Li R, Wang M, Yang D, Liu D, et al. Prophylactic fat-free diet in patients undergoing lobectomy for lung cancer does not decrease postoperative chylothorax: Results from a single-center retrospective study. J BUON 2020;25:1753-60.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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