Challenges of Diagnosis and Management of Congenital Chylothorax in a Low-Resource Setting: A Case Report
DOI:
https://doi.org/10.30442/ahr.1003-11-251Abstract
Chylothorax is a form of pleural effusion. In the neonate, it may be diagnosed in the antenatal or postnatal period. Congenital forms of chylothorax are due to abnormal development or obstruction of the lymphatic system and may be associated with hydrops foetalis; it may also be idiopathic or associated with chromosomal anomalies. The diagnosis is usually based on biochemical analysis of pleural aspirate, and the interpretation of the biochemical parameters depends on whether the baby is feeding. The treatment involves dietary modification, the use of different types of drugs, chest tube drainage, pleurodesis or some other surgical intervention. There are no clinical trials to document the most effective treatment modality. Intrauterine interventions have been reported in some cases. We report a case of neonatal chylothorax to document the diagnostic challenges and management outcomes in a resource-limited setting.
References
Borasino S, Diaz F, El Masri K, Dabal RJ, Alten JA. Central Venous Lines Are a Risk Factor for Chylothorax in Infants After Cardiac Surgery. World J Pediatr Congenit Heart Surg 2014; 5: 522–526. DOI:10.1177/2150135114550723.
Bauman ME, Moher C, Bruce AK, Kuhle S, Kaur S, Massicotte MP. Chylothorax in children with congenital heart disease: Incidence of thrombosis. Thromb Res 2013; 132. https://doi.org/10.1016/J.THROMRES.2013.06.014
Rocha G, Arnet V, Soares P, Gomes AC, Costa S, Guerra P, et al. Chylothorax in the neonate—A stepwise approach algorithm. Pediatr Pulmonol 2021;56:3093–3105. https://doi.org/10.1002/PPUL.25601
Attar MA, Donn SM. Congenital chylothorax. Semin Fetal Neonatal Med 2017;22:234–239. https://doi.org/10.1016/j.siny.2017.03.005
DeWitt E, Michalczyk M. Congenital Chylothorax: Common and Uncommon Findings in an Infant with Chylous Effusions. Neonatal Netw 2019;38:357–364. https://doi.org/10.1891/0730-0832.38.6.357
Bialkowski A, Poets CF, Franz AR. Congenital chylothorax: A prospective nationwide epidemiological study in Germany. Arch Dis Child Fetal Neonatal Ed 2015;100:F169–F172. https://doi.org/10.1136/ARCHDISCHILD-2014-307274
Krishnamurthy B, Malhotra A. Congenital chylothorax: Current perspectives and trends. Res Rep Neonatol 2017;7:53–63. https://doi.org/10.2147/RRN.S128703
Resch B, Sever Yildiz G, Reiterer F. Congenital Chylothorax of the Newborn: A Systematic Analysis of Published Cases between 1990 and 2018. Respiration 2022;101:84–96. https://doi.org/10.1159/000518217
Maldonado F, Hawkins FJ, Daniels CE, Doerr CH, Decker PA, Ryu JH. Pleural fluid characteristics of chylothorax. Mayo Clin Proc 2009;84:129–133. https://doi.org/10.4065/84.2.129
Heffner JE. Etiology, clinical presentation, and diagnosis of chylothorax. In: Finlay G (ed) UpToDate. Wolters Kluwer, https://www.uptodate.com/contents/etiology-clinical-presentation-and-diagnosis-of-chylothorax?search=Congenital%20chylothorax&topicRef=16356&source=see_link (2024, accessed 14 September 2024).
Agrawal V, Doelken P, Sahn SA. Pleural fluid analysis in chylous pleural effusion. Chest 2008;133:1436–1441. https://doi.org/10.1378/CHEST.07-2232
Liou DZ, Warren H, Maher DP, Soukiasian HJ, Melo N, Salim A, et al. A novel therapeutic for refractory chylothorax. Chest 2013;144:1055–1057. https://doi.org/10.1378/CHEST.12-3081
Kasdallah N, Kbaier H, Ben Salem H, Blibech S, Bouziri A, Douagi M. Povidone Iodine Pleurodesis for Refractory Congenital Chylothorax: A Review of Literature. Tunis Med 2016;94:834.
Borcyk K, Kamil A, Hagerty K, Deer M, Tomich P, Anderson Berry AL. Successful management of extremely high-output refractory congenital chylothorax with chemical pleurodesis using 4% povidone-iodine and propranolol: a case report. Clin Case Rep 2018;6:702–708. https://doi.org/10.1002/ccr3.1449
Lee C-J, Tsao P-N, Chen C-Y, Hsieh W-S, Liou J-Y, Chou H-C. Prenatal Therapy Improves the Survival of Premature Infants with Congenital Chylothorax. Pediatr Neonatol 2015;57:127–132. https://doi.org/10.1016/j.pedneo.2015.07.001
Das A, Shah PS. Octreotide for the treatment of chylothorax in neonates. Cochrane Database Syst Rev 2010. https://doi.org/10.1002/14651858.CD006388.PUB2
Bellini C, Cabano R, De Angelis LC, Bellini T, Calevo MG, Gandullia P, et al. Octreotide for congenital and acquired chylothorax in newborns: A systematic review. J Paediatr Child Health 2018;54:840–847. https://doi.org/10.1111/jpc.13889
Weidner WA, Steiner RM. Roentgenographic demonstration of intrapulmonary and pleural lymphatics during lymphangiography. Radiology 1971;100:533–539. https://doi.org/101148/1003533
Xi M, Na X, Ma X, Lan H, Sun T, Liu WH, et al. Maternal diet associated with infants’ intestinal microbiota mediated by predominant long-chain fatty acid in breast milk. Front Microbiol 2023;13: 1004175. https://doi.org/10.3389/FMICB.2022.1004175/BIBTEX
Gupta D, Ross K, Piacentino V, Stepnowski D, McClurken JB, Furukawa S, et al. Use of LeVeen pleuroperitoneal shunt for refractory high-volume chylothorax. Annals of Thoracic Surgery 2004;78:e9-e12. https://doi.org/10.1016/j.athoracsur.2003.12.069
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