Home Print this page Email this page Small font sizeDefault font sizeIncrease font size
Users Online: 135
 
About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Advertise Login 
     


 
 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 6  |  Issue : 3  |  Page : 208-210

Congenital chylothorax in a late preterm neonate associated with hydrops fetalis and successful treatment with octreotide and pleurodesis with betadine


Department of Neonatology, Nice Children Hospital, Bhavnagar, Gujarat, India

Date of Web Publication11-Jul-2017

Correspondence Address:
Hiral Mangukiya
Nice Children Hospital, Sameep Complex, 1st Floor, Opposite Kalubha Bus Stop, Bhavnagar - 364 001, Gujarat
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcn.JCN_47_17

Rights and Permissions
  Abstract 

Congenital chylothorax is an accumulation of chyle in the pleural space that may present in neonatal period with respiratory distress. A 35-week preterm who presented with massive congenital chylothorax complicated with hydrops fetalis. The neonate was treated successfully by total parenteral nutrition, octreotide, and pleurodesis with betadine. Pleurodesis with betadine seems to be effective in the treatment of congenital chylothorax.

Keywords: Chylothorax, hydrops, octreotide, pleurodesis


How to cite this article:
Vaghela PC, Mangukiya H. Congenital chylothorax in a late preterm neonate associated with hydrops fetalis and successful treatment with octreotide and pleurodesis with betadine. J Clin Neonatol 2017;6:208-10

How to cite this URL:
Vaghela PC, Mangukiya H. Congenital chylothorax in a late preterm neonate associated with hydrops fetalis and successful treatment with octreotide and pleurodesis with betadine. J Clin Neonatol [serial online] 2017 [cited 2020 Oct 19];6:208-10. Available from: https://www.jcnonweb.com/text.asp?2017/6/3/208/210144


  Introduction Top


Chylothorax is defined as the accumulation of lymphatic fluid or chyle in the pleural space. The diagnosis of chylothorax is considered when pleural fluid assay has a triglyceride level >1.1 mmol/L (97 mg/dl) and total cell count of >1000 cells/ml with >80% lymphocytes [Figure 1]. Chylothorax can be unilateral or bilateral and congenital or acquired. Congenital chylothorax is associated with abnormalities of the lymphatic system such as lymphangiomatosis and lymphangiectasia, congenital heart disease, chromosomal abnormalities (trisomy 21, Turner's and Noonan syndromes), and H-type of tracheoesophageal fistula). Acquired chylothorax is usually due to trauma to the thoracic duct during cardiac or thoracic surgery.[1] The incidence of congenital chylothorax is reported to be 1 in 10000 births. Many cases of chylothorax have no clear etiology and are considered as idiopathic congenital chylothorax. The reported case fatality rate is 15%–57%; however, mortality is higher when there is associated hydrops fetalis.
Figure 1: Chest X-ray of patient showing bilateral pleural effusion after birth

Click here to view



  Case Report Top


This female neonate was delivered at 35 weeks of gestation to a primigravida mother by means of vaginal delivery. The birth weight was 2555 g, and the Apgar scores were 1 at 2 min and 4 at 5 min. The parents were nonconsanguineous, and bilateral severe pleural effusion and polyhydramnios were detected by routine prenatal ultrasound. After delivery, the neonate was intubated because of poor spontaneous respiratory effort and admitted to the neonatal intensive care unit. The patient had generalized edema but no dysmorphic features. A chest X-ray revealed severe bilateral whiteout of the lung field and bilateral pleural effusion was confirmed by ultrasound.

Bilateral pleural fluid tapping was done and milky white fluid 175 ml and 25 ml from right and left was drained, respectively. Pleural fluid examination showed high leukocytes count. After 6 h, repeat X-ray and ultrasound thorax showed still pleural fluid on right side. Pleural tapping was done and 40 ml fluid was drained.

In view of pleural effusion on right Side, computed tomography scan thorax was done that revealed marked right side pleural effusion with collapse of lung and contralateral tracheal and mediastinal shift. Mild left side pleural effusion with few soft patchy opacity.

Chest tube insertion was performed immediately because marked pleural effusion 70 ml fluid was collected. The drained pleural effusion revealed milky white fluid. The cell counts of the pleural effusion revealed the following: white blood cells 16,000 cells/mm 3 with 80% lymphocytes. Biochemical analysis of the pleural effusion revealed the following: glucose, 84.6 mg/dL; total protein, 2.6 g/dL; lactate dehydrogenase, 263 IU/L; total cholesterol, 47 mg/dL; and triglyceride 220 mg/dl. The albumin level of the plasma was 3.1 g/dL. Total parenteral nutrition (TPN) was initiated.

As there is still persistent pleural fluid drainage through chest tube medical management with octreotide, we started IV infusion of 1 μg/kg/h, and TPN was started and pleural fluid volume has decreased, but it was still persist after 10 days of octreotide and TPN, so pleurodesis with betadine 10% was considered. Betadine 10% was diluted with normal saline (3 ml betadine 10% + 7 ml saline) was instilled through the chest tube. The chest tube was occluded transiently. After 72 h, fluid was significantly reduced, and infant was weaned off to nasal ventilator support. After the commencement of octreotide and pleurodesis, right-side pleural effusion gradually decreased, along with respiratory improvement. The pleural effusion was no longer detectable by ultrasound. After the removal of the chest tubes, ultrasound was performed daily, and no reaccumulation of pleural effusion was detectable.

After treatment with octreotide and pleurodesis, the chylothorax subsided. We could not exclude the possibility that the improvement of chylothorax was because of natural history of the disease [Figure 2].
Figure 2: Follow-up X-ray chest after pleurodesis and octreotide infusion

Click here to view



  Discussion Top


The strategy of treating chylothorax is the same regardless of the etiology of chylothorax. The first step is the aspiration of the pleural fluid for initial drainage and diagnostic purpose. However, continuous drainage of the chyle with a chest tube is indicated if the effusion causes respiratory distress or the accumulation of effusion recurs. Chest tube may be required for a period of time because it takes time for chyle leakage to heal. However, long-term insertion of chest tube has been reported to be associated with hypoproteinemia, lymphopenia, hypogammaglobulinemia, infection, and prolonged ventilator use and its associated lung injury, leading to prolonged hospitalization and emotional stress to the family.[2]

In an attempt to decrease the morbidity usually associated with the classical management of congenital chylothorax (infections, thrombosis, deficit in lymphocytes, deficit in proteins and immunoglobulins, complications of TPN, complications of mechanical ventilation), we treated neonates by intrapleural instillation of povidone-iodine through a chest tube and octreotide.[3]

Octreotide, a synthetic somatostatin analog, is more potent in inhibiting endocrine system and has a much longer half-life. In gastrointestinal tract, somatostatin and octreotide act on somatostatin receptors to reduce intestinal blood flow by vasoconstriction of the splanchnic vessels, decrease gastrointestinal motility, and inhibit gastric, pancreatic, and biliary secretions, thus reducing intestinal fat absorption and lymphatic flow in the thoracic duct. It has also been used for the treatment of congenital chylothorax in term neonates. The experience of octreotide use in premature infants with congenital chylothorax is limited. There is no consensus on the route, dosage, and duration of octreotide administration for chylothorax.

Octreotide is generally considered to be safe, with only occasional side effects. The side effects of octreotide are mainly related to its vasoconstrictive and antisecretory actions. The reported adverse reactions include cramps, flatulence, nausea, diarrhea, necrotizing enterocolitis, hyperglycemia, transient hypothyroidism, and liver dysfunction. No fore mentioned side effects of octreotide were observed in our patient.[4]

In conclusion, octreotide in the treatment of a premature infant with congenital chylothorax appears to be a safe and effective adjunct therapy. It prevented our patient from undergoing reinsertion of chest tubes or surgery. However, further studies in a larger number of patients are required to determine the effectiveness, proper dosing, and side effects of this drug in the treatment of congenital chylothorax.

Neonatal management of congenital chylothorax

Conservative approach

  • Management of underlying disease
  • Repeated thoracocentesis
  • Continuous drainage
  • Dietary modifications (medium chain triacylglycerol diet or TPN)
  • Chest tube pleurodesis
  • Chemical or mechanical pleurodesis
  • Surgical approach
  • Thoracoscopic pleurodesis
  • Pleuroperitoneal pump
  • Surgical abrasion
  • Ligation of the thoracic duct (by thoracoscopy or thoracotomy)
  • Thoracic duct to azygos vein anastomosis and lung transplantation (lymphangioleiomyomatosis).[5]



  Conclusion Top


This report illustrates the feasibility of intrapleural injection of povidone-iodine and octreotide for the treatment of congenital idiopathic chylothorax. This treatment was administered when the classical management of the disease (nil by mouth, parenteral nutrition, pleural punctures, and chest tube drainage) failed in an attempt to avoid more invasive surgical procedures. A recent study confirms that this method has the potential to reduce the morbidity related to prolonged hospital stay. Use of adequate analgesia is a priority.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
van Straaten HL, Gerards LJ, Krediet TG. Chylothorax in the neonatal period. Eur J Pediatr 1993;152:2-5.  Back to cited text no. 1
[PUBMED]    
2.
Beghetti M, La Scala G, Belli D, Bugmann P, Kalangos A, Le Coultre C. Etiology and management of pediatric chylothorax. J Pediatr 2000;136:653-8.  Back to cited text no. 2
    
3.
Wasmuth-Pietzuch A, Hansmann M, Bartmann P, Heep A. Congenital chylothorax: Lymphopenia and high risk of neonatal infections. Acta Paediatr 2004;93:220-4.  Back to cited text no. 3
    
4.
Dubin PJ, King IN, Gallagher PG. Congenital chylothorax. Curr Opin Pediatr 2000;12:505-9.  Back to cited text no. 4
    
5.
Soto-Martinez M, Massie J. Chylothorax: Diagnosis and management in children. Paediatr Respir Rev 2009;10:199-207.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2]



 

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

 
  In this article
Abstract
Introduction
Case Report
Discussion
Conclusion
References
Article Figures

 Article Access Statistics
    Viewed2178    
    Printed14    
    Emailed0    
    PDF Downloaded137    
    Comments [Add]    

Recommend this journal