TY - JOUR T1 - Unusual case of chylothorax with unilateral limb swelling JF - BMJ Case Reports JO - BMJ Case Reports DO - 10.1136/bcr-2019-230379 VL - 13 IS - 2 SP - e230379 AU - Vishal Jha AU - Aditi Jha Y1 - 2020/02/01 UR - http://casereports.bmj.com/content/13/2/e230379.abstract N2 - Here, we present an unusual case of 26-month male toddler who presented with swelling of right lower limb with painless hyperpigmented patch over right groin of 18 months duration associated with recent onset respiratory distress. Evaluation revealed right chylothorax and MRI revealed altered signal intensity in bones and muscles of right lower limb. Lymphoscintigraphy revealed absence of lymphatic channels in right lower limb. Skin biopsy from hyperpigmented patch was suggestive of vasoformative lesion favouring lymphangiomatosis. A diagnosis of Gorham’s syndrome was made, and our patient was managed with drainage of chylothorax followed by pleurodesis, parenteral nutrition and radiotherapy. ER -