Congenital chylothorax in infants: An updated scoping review
Article Sidebar
Main Article Content
Abstract
Chylothorax is a medical condition characterised by the abnormal accumulation of lymphatic fluid in the pleural cavity. In its congenital form, chylothorax develops as a result of an idiopathic abnormality in the thoracic duct. Congenital chylothorax (CCT) is the leading cause of pleural effusion in infants, a condition where excess fluid builds up in the space between the lungs and the chest wall. This scoping review includes a wide range of published studies from 1980 to January 2024, obtained from multiple reputable databases, including Google Scholar, PubMed, Springer and BioMed Central. Effective management strategies are crucial for improving outcomes in infants with CCT. Timely drainage of pleural fluid is essential to alleviate respiratory distress and prevent further complications. Appropriate nutritional care is also critical, as it helps in supporting the neonate’s overall health and recovery. Early intervention and continuous monitoring can significantly improve the likelihood of a positive outcome.
Downloads
Article Details
Issue
Section
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
Authors retain copyright and grant the journal right of first publication.
How to Cite
References
1. Alhasoon MA. The use of high dose octreotide in management of neonatal chylothorax. J Neonatal Perinatal Med 2021;14(4):457-461. https://doi.org/10.3233/ NPM-200644
2. Resch B, Sever Yildiz G, Reiterer F. Congenital chylothorax of the newborn: A systematic analysis of published cases between 1990 and 2018. Respiration 2022;101(1):84-96. https://doi.org/10.1159/000518217
3. Van Straaten HL, Gerards LJ, Krediet TG. Chylothorax in the neonatal period. Eur J Pediatr 1993;152(1):2-5. https://doi.org/10.1007/BF02072505
4. Andrade M, Jacomo A. Anatomy of the human lymphatic system. Cancer Treatment Res 2007;135:55-77. https://doi.org/10.1007/978-0-387-69219-7_5
5. Nastiti PH, Sekarsari D, Setyaningrum RA, et al. Chylothorax: Approaches to diagnosis and treatment. Int J Social Health 2024;3(1):70-81. https://doi.org/10.58860/ ijsh.v3i1.154
6. Duletzke NT, Kiraly LN, Martindale RG. Chylothorax and chylous ascites: Overview, management, and nutrition. Nutr Clin Pract 2023;38(3):557-563. https://doi. org/10.1002/ncp.10973
7. Phang K, Bowman M, Phillips A, Windsor J. Review of thoracic duct anatomical variations and clinical implications. Clin Anat 2014;27(4):637-644. https://doi. org/10.1002/ca.22337
8. HemattiH,MehranRJ.Anatomyofthethoracicduct.ThoracSurgClin2011;21(2):229- 238. https://doi.org/10.1016/j.thorsurg.2011.01.002
9. Song YB. Thoracic duct, cisterna chyli, and right lymphatic duct. In: Tubbs RS, Shoja MM, Loukas M, eds. Bergman’s Comprehensive Encyclopedia of Human Anatomic Variation. 3rd ed. John Wiley & Sons, 2016:921-934.
10. Soto-Martinez M, Massie J. Chylothorax: Diagnosis and management in children. Paediatr Respir Rev 2009;10(4):199-207. https://doi.org/10.1016/j.prrv.2009.06.008
11. Radlinsky M. Lymphatics and lymph nodes management of chylothorax. In: Bojrab MJ, Waldron DR, Toombs JP, eds. Current Techniques in Small Animal Surgery. 5th ed. New York: Teton NewMedia, 2014:671-677.
12. Cacciaguerra S, Catalano P, Antonelli E, Arena S. Pneumothorax and chylothorax. In: Lima M, Reinberg O, eds. Neonatal Surgery: Contemporary Strategies from Fetal Life to the First Year of Age. Cham, Switzerland: Springer, 2019:151-166.
13. Bhatnagar M, Fisher A, Ramsaroop S, Carter A, Pippard B. Chylothorax: Pathophysiology, diagnosis, and management – a comprehensive review. J Thorac Dis 2024;16(2):1645-1661. https://doi.org/10.21037/jtd-23-1636
14. Yu DX, Ma XX, Wang Q, Zhang Y, Li CF. Morphological changes of the thoracic duct and accessory lymphatic channels in patients with chylothorax: Detection with unenhanced magnetic resonance imaging. Euro Radiol 2013;23(3):702-711. https:// doi.org/10.1007/s00330-012-2642-8
15. Kinmonth JB, Taylor GW, Harper RK. Lymphangiography: A technique for its clinical use in the lower limb. Br Med J 1955;1(4919):940-942. https://doi.org/10.1136/ bmj.1.4919.940
16. Kramer EL. Lymphoscintigraphy: Defining a clinical role. Lymphat Res Biol 2004;2(1):32-37. https://doi.org/10.1089/1539685041690454
17. Skouras V, Kalomenidis I. Chylothorax: Diagnostic approach. Curr Opin Pulm Med 2010;16(4):387-393. https://doi.org/10.1097/MCP.0b013e328338dde2
18. Tutor JD. Chylothorax in infants and children. Pediatrics 2014;133(4):722-733. https:// doi.org/10.1542/peds.2013-2072
19. Lopez-Gutierrez JC, Tovar JA. Chylothorax and chylous ascites: Management and pitfalls. Semin Pediatr Surg 2014;23(5):298-302. https://doi.org/10.1053/j. sempedsurg.2014.09.011
20. Mitanchez D, Walter-Nicolet E, Salomon R, Bavoux F, Hubert P. Congenital chylothorax: What is the best strategy? Arch Dis Child Fetal Neonatal Ed 2006;91(2):F153-F154. https://doi.org/10.1136/adc.2004.069690
21. Höck M, Höller A, Hammerl M, et al. Dietary treatment of congenital chylothorax with skimmed breast milk. Ital J Pediatr 2021;47(1):175. https://doi.org/10.1186/ s13052-021-01125-1
22. DeWittE,MichalczykM.Congenitalchylothorax:Commonanduncommonfindings in an infant with chylous effusions. Neonatal Netw 2019;38(6):357-364. https://doi. org/10.1891/0730-0832.38.6.357
23. Bender B, Murthy V, Chamberlain RS. The changing management of chylothorax in the modern era. Eur J Cardiothorac Surg 2016;49(1):18-24. https://doi.org/10.1093/ ejcts/ezv041
24. Wang B, Feng Y, Guo Y, et al. Clinical features and outcomes of congenital chylothorax: A single tertiary medical center experience in China. J Cardiothorac Surg 2022;17(1):276. https://doi.org/10.1186/s13019-022-02009-z
25. Attar MA, Donn SM. Congenital chylothorax. Semin Fetal Neonatal Med 2017;22(4):234-239. https://doi.org/10.1016/j.siny.2017.03.005
26. Krishnamurthy MB, Malhotra A. Congenital chylothorax: Current perspectives and trends. Res Rep Neonatol 2017;7:53-63. https://doi.org/10.2147/RRN.S128703
27. Panthongviriyakul C, Bines JE. Post-operative chylothorax in children: An evidence- based management algorithm. J Paediatr Child Health 2008;44(12):716-721. https:// doi.org/10.1111/j.1440-1754.2008.01412.x
28. Abuhamda AF, Elsous A, Al-Saadi N, Al-Dadah M. A rare case of congenital chylothorax in a Palestinian neonate. Respir Med Case Rep 2019;28:100937. https:// doi.org/10.1016/j.rmcr.2019.100937
29. Oh JH, Lee G, Lee JH. Congenital chylothorax and pulmonary hypertension complicated with heart failure and hepatopathy. Pediatr Int 2013;55(2):e7-e10. https:// doi.org/10.1111/j.1442-200X.2012.03700.x
30. Muniz G, Hidalgo-Campos J, Valdivia-Tapia MD, Shaikh N, Carreazo NY. Successful management of chylothorax with etilefrine: Case report in 2 pediatric patients. Pediatrics 2018;141(5):e20163309. https://doi.org/10.1542/peds.2016-3309
31. Tomobe Y, Mizuguchi U, Shimotakahara A, Shimojima N, Okazaki K. Combination therapy with etilefrine and pleurodesis for refractory congenital chylothorax. Biomed Hub 2020;5(3):1-5. https://doi.org/10.1159/000509903
32. Tamaoka S, Osada A, Kin T, Arimitsu T, Hida M. Midodrine, an oral alpha-1 adrenoreceptor agonist, successfully treated refractory congenital chylous pleural effusion and ascites in a neonate. Chest 2021;159(4):e189-e191. https://doi. org/10.1016/j.chest.2020.10.071
33. Malleske DT, Yoder BA. Congenital chylothorax treated with oral sildenafil: A case report and review of the literature. J Perinatol 2015;35(5):384-386. https://doi. org/10.1038/jp.2015.10
34. UzodimmaCC,SanniSB,Madise-WoboDA,UzodimmaCC.Congenitalchylothorax treated with oral sildenafil – case report from Nigeria and review of literature. Open J Pediatr 2023;13(2):189-195. https://doi.org/10.4236/ojped.2023.132024
35. Bhagiratha S, Bandiya P, Shivanna N, Benakappa N. Congenital chylothorax: Conservative management counts. J Indian Assoc Pediatr Surg 2022;27(5):652-654. https://doi.org/10.4103/jiaps.jiaps_256_21
36. Handal-OreficeR,MiduraD,WuJK,ParraviciniE,MillerRS,ShawberCJ.Propranolol therapy for congenital chylothorax. Pediatrics 2023;151(2):e2022058555. https://doi. org/10.1542/peds.2022-058555
37. Mitchell K, Weiner A, Ramsay P, Sahni M. Use of propranolol in the treatment of chylous effusions in infants. Pediatrics 2021;148(1):e2020049699. https://doi. org/10.1542/peds.2020-049699
38. Liviskie CJ, Brennan CC, McPherson CC, Vesoulis ZA. Propranolol for the treatment of lymphatic malformations in a neonate – a case report and review of literature. J Pediatr Pharmacol Ther 2020;25(2):155-162. https://doi.org/10.5863/1551-6776- 25.2.155
39. SersarSI.Predictorsofprolongeddrainageofchylothoraxaftercardiacsurgery:Single centre study. Pediatr Surgery Int 2011;27(8):811-815. https://doi.org/10.1007/s00383- 011-2890-8
40. FujinoS,MaruyamaH,TsukamotoK,OnoH,IsayamaT,ItoY.Chylothoraxassociated with congenital complete atrioventricular block. AJP Rep 2020;10(04):e403-e407. https://doi.org/10.1055/s-0040-1715178
41. Graham DD, McGahren ED, Tribble CG, Daniel TM, Rodgers BM. Use of video- assisted thoracic surgery in the treatment of chylothorax. Ann Thoracic Surg 1994;57(6):1507-1512. https://doi.org/10.1016/0003-4975(94)90110-4
42. Allman DA, Radlinsky MG, Ralph AG, Rawlings CA. Thoracoscopic thoracic duct ligation and thoracoscopic pericardectomy for treatment of chylothorax in dogs. Vet Surg 2010;39(1):21-27. https://doi.org/10.1111/j.1532-950X.2009.00623.x
43. Samanidis G, Kourelis G, Bounta S, Kanakis M. Postoperative chylothorax in infants and infants after congenital heart disease surgery – current aspects in diagnosis and treatment. Nutrients 2022;14(9):1803. https://doi.org/10.3390/nu14091803
44. Mahenthiran AK, Brajcich BC, Kim S, Bharat A, Odell DD. National assessment of early discharge after video-assisted thoracoscopic surgery for lung resection. J Surg Res 2022;276:242-250. https://doi.org/10.1016/j.jss.2022.02.025
45. Liu CY, Hsu PK, Huang CS, Sun YH, Wu YC, Hsu WH. Chylothorax complicating video-assisted thoracoscopic surgery for non-small cell lung cancer. World J Surg 2014;38(11):2875-2881. https://doi.org/10.1007/s00268-014-2699-4
46. Healy H, Gipson K, Hay S, Bates S, Kinane TB. Management and outcomes of congenital chylothorax in the neonatal intensive care unit: A case series. Pediatr Investig 2017;1(1):21-25. https://doi.org/10.1002/ped4.12007
47. Li CW, Chiang MC, Lien R, Tsai TC, Hsu KH. Sildenafil to treat congenital chylothorax: The first case report in Taiwan. Pediatr Neonatol 2024;65(1):98-100. https://doi.org/10.1016/j.pedneo.2023.06.006
48. Resch B, Halmer M, Müller WD, Eber E. Long-term follow-up of children with congenital chylothorax. Eur Respir J 2012;40(4):1060-1062. https://doi. org/10.1183/09031936.00016812
49. Vain NE, Swarner OW, Cha CC. Neonatal chylothorax: A report and discussion of nine consecutive cases. J Pediatr Surg 1980;15(3):261-265. https://doi.org/10.1016/ s0022-3468(80)80133-3
50. Resch B. Management of congenital chylothorax of the newborn. Respiration 2022;101(8):795-796. https://doi.org/10.1159/000525377
51. Kim JH, Katheria A. Clinical syndromes and associations with persistent pulmonary hypertension of the newborn. In: Sulica R, Preston I, eds. Pulmonary Hypertension – From Bench Research to Clinical Challenges. InTech, 2011. https://doi. org/10.5772/26728
52. Al-Tawil K, Ahmed G, Al-Hathal M, Al-Jarallah Y, Campbell N. Congenital chylothorax. Am J Perinatol 2000;17(3):121-126. https://doi.org/10.1055/s-2000-9281 53. Porcel JM, Azzopardi M, Koegelenberg CF, Maldonado F, Rahman NM, Lee YC. The diagnosis of pleural effusions. Exp Rev Respir Med 2015;9(6):801-815. https://doi.or
g/10.1586/17476348.2015.1098535
43. Porcel JM, Azzopardi M, Koegelenberg CF, Maldonado F, Rahman NM, Lee YC. The diagnosis of pleural effusions. Expert review of respiratory medicine. 2015 Nov 2;9(6):801-15.