{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T03:22:56Z","timestamp":1778037776667,"version":"3.51.4"},"reference-count":29,"publisher":"BMJ","issue":"2","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2020,1,27]]},"published-print":{"date-parts":[[2020,2]]},"abstract":"<jats:p>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\u2019s syndrome was made, and our patient was managed with drainage of chylothorax followed by pleurodesis, parenteral nutrition and radiotherapy.<\/jats:p>","DOI":"10.1136\/bcr-2019-230379","type":"journal-article","created":{"date-parts":[[2020,2,11]],"date-time":"2020-02-11T04:19:16Z","timestamp":1581394756000},"page":"e230379","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":5,"title":["Unusual case of chylothorax with unilateral limb swelling"],"prefix":"10.1136","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2244-1651","authenticated-orcid":false,"given":"Vishal","family":"Jha","sequence":"first","affiliation":[{"name":"Department of Medicine, Army Hospital Research and Referral, New Delhi, Delhi, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3961-411X","authenticated-orcid":false,"given":"Aditi","family":"Jha","sequence":"additional","affiliation":[{"name":"Department of Anaesthesiology, General Hospital, Ladakh, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2020,2,10]]},"reference":[{"key":"2025082802132498000_13.2.e230379.1","doi-asserted-by":"publisher","DOI":"10.1016\/0046-8177(93)90265-I"},{"key":"2025082802132498000_13.2.e230379.2","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm.161.3.9904056"},{"key":"2025082802132498000_13.2.e230379.3","doi-asserted-by":"publisher","DOI":"10.1089\/lrb.2011.0023"},{"key":"2025082802132498000_13.2.e230379.4","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-199304000-00002"},{"key":"2025082802132498000_13.2.e230379.5","doi-asserted-by":"publisher","DOI":"10.3121\/cmr.3.2.65"},{"key":"2025082802132498000_13.2.e230379.6","doi-asserted-by":"crossref","first-page":"88","DOI":"10.1302\/0301-620X.67B1.3968152","article-title":"The syndrome of idiopathic osteolysis. 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