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Table of Contents
CASE REPORT
Year : 2022  |  Volume : 12  |  Issue : 4  |  Page : 196-199

Postoperative chylothorax and left internal jugular vein thrombus after complex cyanotic heart disease surgery


Department of Pediatrics, SMS Medical College, Jaipur, Rajasthan, India

Date of Submission04-Aug-2022
Date of Decision19-Aug-2022
Date of Acceptance23-Aug-2022
Date of Web Publication19-Dec-2022

Correspondence Address:
Dr. Dhan Raj Bagri
Sir Padampat Institute of Neonatology and Child Health, J K Lone Hospital, SMS Medical College, Jaipur, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jicc.jicc_26_22

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  Abstract 


Chylothorax is the accumulation of lymphatic fluid in pleural space following traumatic injury to lymphatic vessels, systemic venous obstruction, dysfunction of the right ventricle, thrombosis of the duct, superior vena cava or subclavian vein, or postoperatively. A 2 ½-year-old male child operated for transposition of great arteries (TGA), tricuspid atresia (TA), and hypoplastic right ventricle 15 days ago developed chylothorax and left internal jugular vein thrombus. The child was initially managed conservatively with nutritional management, anticoagulation, and octreotide followed by interventional radiological management with lipoidol injection. Early suspicion and diagnosis are crucial. The management was difficult owing to a lack of proper guidelines. Further research is warranted.

Keywords: Complex cyanotic heart disease surgery, left internal jugular vein thrombus, lipiodol, postoperative chylothorax


How to cite this article:
Bagri DR, Meena K, Meena JS, Gurjar U, Jeengar B. Postoperative chylothorax and left internal jugular vein thrombus after complex cyanotic heart disease surgery. J Indian coll cardiol 2022;12:196-9

How to cite this URL:
Bagri DR, Meena K, Meena JS, Gurjar U, Jeengar B. Postoperative chylothorax and left internal jugular vein thrombus after complex cyanotic heart disease surgery. J Indian coll cardiol [serial online] 2022 [cited 2023 Feb 8];12:196-9. Available from: https://www.joicc.org/text.asp?2022/12/4/196/364214




  Introduction Top


Chylothorax is the accumulation of lymphatic fluid in pleural space following traumatic injury to lymphatic vessels, systemic venous obstruction, dysfunction of the right ventricle, thrombosis of the duct, superior vena cava (SVC) or subclavian vein, or postoperative chylothorax. The Pediatric Cardiac Critical Care Consortium (PC4) and the Pediatric Health Information System databases suggest a 2.8%–3.8% incidence of chylothorax in pediatric patients following congenital heart surgery or heart transplantation, with more incidence (6.9%) in patients with single-ventricle physiology.[1],[2] Atriopulmonary and cavopulmonary anastomosis (Glenn shunt or Fontan operation) may lead to damage to the thoracic duct or collateral lymphatics during dissection or SVC cannulation and have a higher incidence (5.7%) of postoperative chylothorax.[3]


  Case Report Top


A 2½-year-old male child with a weight of 8 kg, presented to the emergency department in low general condition with complaints of fever, swelling over the face, neck, and chest and respiratory distress, cyanosis, and bilateral crepitations on the 15th postoperative day. He was operated on for TGA, TA, and hypoplastic right ventricle 15 days ago and a bidirectional Glenn procedure was done with cardiac surgery at another institute. He was discharged on the 6th postoperative day.

On the 8th postoperative day, he developed swelling over the face, neck, and chest and difficulty in breathing. The child was consulted at a tertiary center emergency on the 12th day of surgery and a provisional diagnosis of cyanotic CHD (tricuspid atresia, mispositioned great arteries, and hypoplastic right ventricle operated with BD Glenn procedure) with postoperative right-sided pleural empyema, or chylothorax. An intercostal drainage tube was placed and after stabilization referred to our center on the 15th postoperative day [Figure 1].
Figure 1: Chylothorax

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His heart rate was 134/min, respiratory rate was 66/min, and air entry was diminished on the right side; bilateral crepitations were audible with cyanosis (SpO2 – 84%), subcostal, and intercostal retractions. A healing scar mark was present on the chest wall in the sternal area. Intercostal Drainage Tube (ICD) was in situ and drained yellowish white fluid which suggested chyle/superadded infection. Owing to a history of cardiac surgery 15 days ago and right-sided empyema; a Cardiothoracic and Vascular Surgery (CTVS) reference was done, which suggested no active surgical intervention was required at that time and advised stabilization and review.

Investigations [Table 1] – Pleural fluid was grossly pale white, cell counts suggested white blood cell (WBC) 1100 cells/mm3, with polymorphs – 20%, lymphocytes – 80%, and red blood cell – nil. Pleural fluid gram staining suggested – no pus cells; few gram-positive cocci in chains were noted. Pleural fluid biochemistry is depicted in [Table 2]. Pleural fluid culture sensitivity detected Burkholderia cepacia which was sensitive to ceftazidime, gentamicin, minocycline, chloramphenicol, and levofloxacin.
Table 1: Routine blood investigations

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Table 2: Pleural fluid biochemistry

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Arterial blood gas analysis was normal. Hepatitis B surface antigen, HIV, and HCV were negative. The blood culture and sensitivity report were negative for pathogenic bacteria. Ultrasound Doppler of the neck suggested long segment thrombus extending from left internal jugular vein (IJV) to inferior vena cava. The right IJV suggested normal flow. Other vessels were normal. Few cervical lymph nodes of levels 2 and 3 were noted. Chest X-ray and ultrasound chest suggested right lung consolidation with air bronchogram and minimal left pleural effusion [Figure 2].
Figure 2: Chest X-ray

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Contrast-enhanced computed tomography (CECT) scan thorax [Figure 3]. SVC is opening into the right pulmonary artery. Left IJV and subclavian vein showed hypodense filling defect extending into SVC. The left ventricle appeared dilated with right-sided aortic arch with surrounding edema involving the neck and anterior mediastinum. Inferior vena cava was draining into the right atrium. Anterior mediastinal vessels appeared engorged with edema. A large ventricular septal defect was noted. Subsegmental collapse with consolidation was noted in the right lower lobe with mild pleural effusion. The tip of the ICD was seen in the right middle lobe parenchyma. Trachea was central and bifurcation and bronchial segmental divisions were normal. No obvious intraluminal pathology was noted. Visualized upper abdominal viscera did not reveal any significant abnormality. Congenital heart disease (TGA with TA) with postoperative consolidation with mild right pleural effusion and ICD in situ with thrombus seen in left IJV, extending up to the SVC with surrounding edema involving the neck and anterior mediastinum was concluded.
Figure 3: CECT Scan Chest

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Management – The child was given broad-spectrum antibiotics for the suspicion of postoperative sepsis. Routine investigations including ultrasound and CECT neck were done on day 2 of admission and CTVS and pediatric surgery opinions were taken. Anticoagulants were started for IJV thrombus. Medium-Chain Triglyceroid (MCT) oil was added to milk for nasogastric feeding. The child was having a drain of 1600 ml of chyle through ICD on admission which was replaced with IVF. Calcium was given and Vitamins A, D, E, and K supplemented. The same amount of the drain continued for 5 days. The coagulation profile was repeated and the dose of warfarin was increased due to an INR of 1.21. Octreotide was started on day 6 and we increased the dose as per protocol we noticed a diminution of ICD fluid quantity on day 12 the child had loose stools and was given zinc, probiotic, and oral rehydration solution (ORS). An interventional radiologist consulted on day 16 for lymphangiography and lipiodol injection, as the drainage fluid was still 300 ml per day. A plain chest, abdomen, and pelvis CT along with routine investigations were advised by an interventional radiologist along with clamping of the ICD. We tapered octreotide and lipiodol was injected into bilateral inguinal lymph nodes by the interventional radiologist. Postlymphangiography evaluation (noncontrast CT scan chest and abdomen) revealed opacification of bilateral inguinal lymph nodes and along the lymphatics reaching up to the level of the upper border of L2 lumbar vertebra, thereafter no contrast opacification was noted.


  Discussion Top


Chylothorax in the postoperative period is usually suspected in cases with milky and persistent chest tube drainage. Pleural fluid analysis, suggesting a WBC count >1000 cells/μL with >70%–80% lymphocytes, and a triglyceride concentration >110 mg/dL in enterally fed children is diagnostic of chylothorax. A diagnostic algorithm is used for establishing the diagnosis.[4]

Treatment is aimed to decrease the thoracic lymph flow and support is continued until the lymphatic vessels heal or develop and lymph flow ceases. Nutritional management is conservative in the form of nil by mouth plus total parenteral nutrition (TPN) or enteral feeding with an MCT diet, fat-modified breast milk, or a low-fat diet. MCT is absorbed from the intestine and directly transported to the portal system without chylomicron formation and may physiologically reduce flow in the thoracic duct.[5] Octreotide (or somatostatin) is a second-line pharmacologic adjunctive treatment to reduce lymph flow. Data regarding its use are limited and no guidelines about the onset of chylothorax and starting of octreotide infusion have been speculated.[5],[6]

There is a lack of guidelines about output classification of chylous effusions as low and high, nutritional management, duration of enteral feeding in high-output states, and optimum care for such cases. Output >10 mL/kg/day is generally considered high. No guidelines address TPN administration in a child already having IJV thrombosis.[7]

If conservative management is not successful in decreasing the chylous output <100 mL/kg/day (or 100 mL/year of age) for 5 days, or chylous output >100 mL/day persistently for >2 weeks, or remains unchanged for 1–2 weeks, surgical management is indicated. Thoracic duct ligation is successful in 90% of cases but has high mortality risks. Takahashi et al. proposed pleurodesis using OK432 as a surgical first-line therapy for chylothorax even for neonates.[8] Thoracic duct embolization with microcatheters using lipiodol has been associated with several complications, such as stroke, pulmonary edema, and lymphedema.[9] Lymphovenous anastomosis has also been tried.[10]


  Conclusions Top


Postoperative chylothorax is a rare complication after congenital heart disease surgery. Early suspicion and diagnosis are important for pediatricians. The definition, classification as low or high output, the indication of nutritional management, duration of conservative management, indications of interventional and surgical management, and role of octreotide should be better researched. A multidisciplinary team including pediatricians, pediatric surgeons, cardiothoracic and vascular surgeons, and interventional radiologists is needed for managing such cases.

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.
Mery CM, Moffett BS, Khan MS, Zhang W, Guzmán-Pruneda FA, Fraser CD Jr., et al. Incidence and treatment of chylothorax after cardiac surgery in children: Analysis of a large multi-institution database. J Thorac Cardiovasc Surg 2014;147:678-86.e1.  Back to cited text no. 1
    
2.
Buckley JR, Graham EM, Gaies M, Alten JA, Cooper DS, Costello JM, et al. Clinical epidemiology and center variation in chylothorax rates after cardiac surgery in children: A report from the Pediatric Cardiac Critical Care Consortium. Cardiol Young 2017;27:1678-85.  Back to cited text no. 2
    
3.
Chan SY, Lau W, Wong WH, Cheng LC, Chau AK, Cheung YF. Chylothorax in children after congenital heart surgery. Ann Thorac Surg 2006;82:1650-6.  Back to cited text no. 3
    
4.
Skouras V, Kalomenidis I. Chylothorax: Diagnostic approach. Curr Opin Pulm Med 2010;16:387-93.  Back to cited text no. 4
    
5.
Aljazairi AS, Bhuiyan TA, Alwadai AH, Almehizia RA. Octreotide use in post-cardiac surgery chylothorax: A 12-year perspective. Asian Cardiovasc Thorac Ann 2017;25:6-12.  Back to cited text no. 5
    
6.
Madhavan S, Nakao M. How efficacious are Octreotide and Somatostatin in the management of chylothorax in congenital cardiac surgical patients? Interact Cardiovasc Thorac Surg 2021;33:773-8.  Back to cited text no. 6
    
7.
Samanidis G, Kourelis G, Bounta S, Kanakis M. Postoperative chylothorax in neonates and infants after congenital heart disease surgery-current aspects in diagnosis and treatment. Nutrients 2022;14:1803.  Back to cited text no. 7
    
8.
Takahashi Y, Kinoshita Y, Kobayashi T, Arai Y, Ohyama T, Yokota N, et al. Management of refractory chylothorax in the neonatal Intensive Care Unit: A 22-year experience. Pediatr Int 2022;64:e15043.  Back to cited text no. 8
    
9.
Savla JJ, Itkin M, Rossano JW, Dori Y. Post-operative chylothorax in patients with congenital heart disease. J Am Coll Cardiol 2017;69:2410-22.  Back to cited text no. 9
    
10.
Weissler JM, Cho EH, Koltz PF, Carney MJ, Itkin M, Laje P, et al. Lymphovenous anastomosis for the treatment of chylothorax in infants: A novel microsurgical approach to a devastating problem. Plast Reconstr Surg 2018;141:1502-7.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1], [Table 2]



 

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