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CASE REPORT
Year : 2023  |  Volume : 28  |  Issue : 1  |  Page : 66-68
 

Evolution of antenatally diagnosed bronchogenic cyst in an infant


1 Department of Neonatology, Pediatric Surgery and Pathology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
2 Department of Pediatrics, Sri Aurobindo Institute of Medical Sciences, Indore, Madhya Pradesh, India

Date of Submission14-May-2021
Date of Decision01-Jul-2022
Date of Acceptance30-Jul-2022
Date of Web Publication10-Jan-2023

Correspondence Address:
Anita Singh
Department of Neonatology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.jiaps_87_21

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   Abstract 


Bronchogenic cyst is one of the rare developmental lung conditions. Depending on the location, it can cause significant compression of the mediastinal structures, especially airways leading to atelectasis, emphysema, wheezing, and stridor. Computerized tomography helps in the confirmation of diagnosis. Surgery is definitive management. We present a case of bronchogenic cyst which presented as emphysema leading to respiratory emergency in an infant.


Keywords: Bronchogenic cyst, infant, respiratory distress
Key Message

  • A small hilar mass may be missed at birth, clinically and radiologically and can later evolve to present as airway obstruction later leading to life-threatening emergency
  • Posterior mediastinal, prevertebral, and pericarinal location of the cyst have a potential for bilateral airway compromise.


How to cite this article:
Singh A, Mandelia A, Kawdiya A, Naranje K, Nigam N. Evolution of antenatally diagnosed bronchogenic cyst in an infant. J Indian Assoc Pediatr Surg 2023;28:66-8

How to cite this URL:
Singh A, Mandelia A, Kawdiya A, Naranje K, Nigam N. Evolution of antenatally diagnosed bronchogenic cyst in an infant. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Feb 2];28:66-8. Available from: https://www.jiaps.com/text.asp?2023/28/1/66/367392





   Introduction Top


Bronchogenic cyst is one of the rare developmental lung malformations which results from abnormal budding or branching of the tracheobronchial tree.[1] It can be diagnosed antenatally either as an intrathoracic fluid filled cystic cavity or echogenic structure in the lung. It can even result in the compression of airway in the fetus necessitating need of ex-utero intrapartum treatment following delivery. The most common location is in the mediastinum and thorax. After birth, it may remain asymptomatic or present with symptoms of airway compression. We present a case of antenatally diagnosed bronchogenic cyst which was asymptomatic at birth but presented later as severe respiratory distress resulting from emphysema due to compression of bronchus.


   Case Report Top


A 2.5 months old female infant presented with complaints of difficulty in breathing for 7 days. At the time of admission, she was in respiratory distress with a respiratory rate of 70 breaths per minute, moderate intercostal and subcostal retractions along with markedly decreased air entry in the left hemithorax. She was tachycardiac (HR 152/min) and hypoxic (Spo2 90% on room air).

She was delivered to a primigravida mother at 334/7 weeks of gestation by emergency caesarean section. Ultrasound at 20 weeks of gestation revealed a uniloculated 0.8 cm × 0.5 cm sized cystic mass with a smooth contour in the left thoracic cavity without any mediastinal shift or decrease in lung volume. No other anomaly was detected. Baby cried immediately after birth and the birth weight was 1920 grams. In the immediate neonatal period, baby had transient respiratory distress soon after birth, for which she required continuous positive airway pressure for 4 hours. The chest X-ray at birth was apparently normal [Figure 1]a. She was discharged on day 4 of life.
Figure 1: (a) Normal chest X-ray at birth; (b) Preoperative CXR at 3 months of age showing emphysema of left side with shift of mediastinum; (c) Postoperative CXR showing decrease in left sided hyperinflation, CXR: Chest X-ray

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In the current presentation, the X-ray chest showed hyperinflation of the left hemithorax with depression of the left hemidiaphragm and widening of rib spaces. There was herniation of the left lung to the right side, causing compression of the right lung along with shifting of mediastinum [Figure 1]b. CT chest showed a posterior mediastinal cyst of size 26 mm × 13 mm × 28 mm, abutting the carina, more toward the left hemithorax, causing deviation of esophagus to the right side and focal attenuation of the left main bronchus. Equal hyperinflation of the left upper and lower lobe of lung were noted [Figure 2]a.
Figure 2: (a) CT chest image showing large posterior mediastinal cyst causing attenuation of left main bronchus (black arrow) with close proximity to right bronchus (yellow arrow) and esophagus (blue arrow); (b) Cyst lined by respiratory type of epithelium consisted of ciliated pseudostratified columnar cells.(H and E, ×200). CT: Computed tomography

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After initial stabilization, diagnosis and localization of cyst in computed tomography (CT) scan, surgery was planned. On left posterolateral thoracotomy, a posterior mediastinal cyst was seen at carina compressing the left main bronchus, resulting in emphysematous left lung. Excision of the posterior mediastinal cyst was done. Postoperatively, she required intensive care support in the form of mechanical ventilation (for 8 days), parenteral nutrition, inotropes, and antibiotics. She further required noninvasive respiratory support for 12 days. The post-operative need for ventilation was augmented by infection and pneumonia. The post-operative X-ray showed a decrease in left-sided hyperinflation [Figure 1]c. The gavage feeding could be started 7 days after surgery and was gradually increased to full feeds followed by cup and spoon feeds. The biopsy revealed a cyst wall lined by cuboidal to psedostratified ciliated columnar lining epithelium, underlying subepithelium showing mucous glands and foci of mature cartilaginous tissue along with bronchial mucosa, suggestive of a bronchogenic cyst [Figure 2]b. Baby remained well in follow-up (till 3 years of age) without any episodes of respiratory tract infection or hospitalization.


   Discussion Top


Bronchogenic cyst constitute approximately 13%–15% of congenital cystic lung diseases and 6% of mediastinal masses in children.[1] The location of the cyst depends on timing of the abnormal budding during embryogenesis. It is usually unilocular and has no communication with the trachea-bronchial tree. If it communicates with the airway, it can present with infection or as a tension cyst. Mediastinum is the most common (85%) location, followed by the lung parenchyma. Unusual locations such as head, neck, pericardium, and abdomen have been reported previously.[2] In the present case, it was in the posterior mediastinum. The mediastinal bronchogenic cysts have been classified in five types by Maier based on their location: paratracheal, carinal, hilar, paraesophageal, and miscellaneous. It can be symptomatic in 70% of cases which is mostly because of compression or infection. Mediastinal cysts usually present with compression effect while intrapulmonary cysts present as infection. Compression is a common presentation in younger age, while infection is more common among adults. One third of the cases may remain asymptomatic and may be detected incidentally. Antenatal diagnosis is possible in 70% of cases.[3] In the present case, the bronchogenic cyst was diagnosed on antenatal ultrasound and presented with compressive symptoms at 2.5 months of age.

The compression of airway can lead to severe respiratory distress in the form of stridor or wheezing. In the present case, the infant was not symptomatic at birth but presented later. The methods of postnatal diagnosis include chest X-ray, CT, barium swallow, and bronchoscopy. Chest radiograph may be normal in 23% of cases.[4] It may show homogenous opacity, deviation of mediastinum or trachea, and effect of compression in form of atelectasis or emphysema. In our case, the chest X-ray showed subtle abnormality at birth. At the age of 2.5 months, there were findings suggestive of airway compression in form of emphysema. Without an antenatal diagnosis, it would have been very difficult to differentiate it from congenital lobar emphysema on an X-ray before doing a diagnostic CT. CT helps in accurate diagnosis, better delineation of anatomy, and to rule out other differential diagnosis. Bronchoscopy may reveal narrowing of the lumen of airways in cases presenting with wheeze and stridor because of compression. Because of potential risk of compression effect and malignant transformation, the resection is indicated even in asymptomatic cases.[5] Simple excision is needed in mediastinal cysts, while intraparenchymal cysts may require segmentectomy or even lobectomy.


   Conclusion Top


Bronchogenic cysts can present with severe respiratory distress in infants and children. The asymptomatic cases should be followed up vigilantly for early identification, diagnosis, and surgical management.

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 initial s 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.
Limaiem F, Mlika M. Bronchogenic cyst. In: StatPearls. Treasure Island (FL): StatPearls Publishing; 2021. p. 10.  Back to cited text no. 1
    
2.
Jiang JH, Yen SL, Lee SY, Chuang JH. Differences in the distribution and presentation of bronchogenic cysts between adults and children. J Pediatr Surg 2015;50:399-401.  Back to cited text no. 2
    
3.
Davenport M, Warne SA, Cacciaguerra S, Patel S, Greenough A, Nicolaides K. Current outcome of antenally diagnosed cystic lung disease. J Pediatr Surg 2004;39:549-56.  Back to cited text no. 3
    
4.
Ryu JH, Swensen SJ. Cystic and cavitary lung diseases: Focal and diffuse. Mayo Clin Proc 2003;78:744-52.  Back to cited text no. 4
    
5.
Cheng L, Duan J, Wang M, Lu D, Li H, Ma J, et al. Case report: Prenatal and postnatal management for fetal bronchogenic cysts during The COVID-19 pandemic. Front Pediatr 2021;9:675883.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2]



 

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