An Unusual Presentation of Spontaneous Chylothorax.

True bilateral spontaneous chylothorax without any etiology has been reported rarely in the pediatric literature. A 3-year-old male child was detected to have incidental moderate chylothorax on USG thorax done for scrotal swelling. Investigations for infectious, malignant, cardiac, and congenital etiology were unremarkable. Effusion was drained by securing bilateral intercostal drains (ICD) and confirmed to be chyle on biochemical evaluation. The child was discharged with ICD in situ, but there was non-resolution of bilateral pleural effusion. Because of the failure of conservative treatment, video-assisted thoracoscopy (VATS) with pleurodesis was done. Thereafter, the child improved symptomatically and was discharged. On follow-up, there is no recurrence of pleural effusion, and the child has been growing well, albeit the etiology remains elusive. Chylothorax should not be missed in children presenting with scrotal swelling. In children with spontaneous chylothorax, VATS should be done after a fair trial of conservative medical management (thoracic drainage) along with continued nutritional management. How to cite this article: Kaul A, Fursule A, Shah S. An Unusual Presentation of Spontaneous Chylothorax. Indian J Crit Care Med 2022;26(7):871-873.