Literature DB >> 24000203

Propranolol as a treatment option in Gorham-Stout syndrome: a case report.

Vered Nir1, Ludmila Guralnik, Galit Livnat, Ronen Bar-Yoseph, Fahed Hakim, Anat Ilivitzki, Lea Bentur.   

Abstract

Gorham-Stout syndrome is a very rare syndrome characterized by progressive angiomatosis and lymphangiomas involving multiple organs. We describe herein a girl with progressive Gorham-Stout syndrome since the age of 13 years. Her disease involved the mediastinum, pericardium, vertebrae, ribs, and skull and did not respond to interferon and bisphosphonates. Propranolol administration was initiated at the age of 18 years and was associated with improvement in pulmonary function tests and involution of the mediastinal and hilar hemangiomatous lesions. The possible beneficial effect of propranolol in Gorham-Stout syndrome should be further investigated.
© 2013 Wiley Periodicals, Inc.

Entities:  

Keywords:  Gorham-Stout syndrome; propranolol

Mesh:

Substances:

Year:  2013        PMID: 24000203     DOI: 10.1002/ppul.22869

Source DB:  PubMed          Journal:  Pediatr Pulmonol        ISSN: 1099-0496


  8 in total

1.  Thoracic involvement of diffuse lymphangiomatosis successfully treated with sildenafil.

Authors:  Arnaud Maurac; Marie-Pierre Debray; Bruno Crestani; Camille Taillé
Journal:  BMJ Case Rep       Date:  2019-04-23

2.  Initial Experience With Propranolol Treatment of Lymphatic Anomalies: A Case Series.

Authors:  June K Wu; Ellen D Hooper; Sherelle L Laifer-Narin; Lynn L Simpson; Jessica Kandel; Carrie J Shawber
Journal:  Pediatrics       Date:  2016-08-25       Impact factor: 7.124

3.  Gorham-Stout disease: radiological, histological, and clinical features of 12 cases and review of literature.

Authors:  Yi Liu; Ding-Rong Zhong; Pei-Ran Zhou; Fang Lv; Dou-Dou Ma; Wei-Bo Xia; Yan Jiang; Ou Wang; Xiao-Ping Xing; Mei Li
Journal:  Clin Rheumatol       Date:  2014-09-18       Impact factor: 2.980

4.  Pregnancy Complicated by Gorham-Stout Disease and Refractory Chylothorax.

Authors:  Jessica Hellyer; Hunter Oliver-Allen; Majid Shafiq; Alisha Tolani; Maurice Druzin; Michael Jeng; Stanley Rockson; Robert Lowsky
Journal:  AJP Rep       Date:  2016-10

Review 5.  Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases.

Authors:  Andrea Angelini; Nicolò Mosele; Elisa Pagliarini; Pietro Ruggieri
Journal:  EFORT Open Rev       Date:  2022-01-11

6.  The therapeutic response in Gorham's syndrome to the beta-blocking agent propranolol is correlated to VEGF-A, but not to VEGF-C or FLT1 expression.

Authors:  Jessica Baud; Abderrahim Lomri; Denis Graber; Andreas Bikfalvi
Journal:  BMC Res Notes       Date:  2015-08-04

7.  Pulmonary and pleural lymphatic endothelial cells from pediatric, but not adult, patients with Gorham-Stout disease and generalized lymphatic anomaly, show a high proliferation rate.

Authors:  Michiko Mori; Michael Dictor; Nicholas Brodszki; Juan Carlos López-Gutiérrez; María Beato; Jonas S Erjefält; Erik A Eklund
Journal:  Orphanet J Rare Dis       Date:  2016-05-18       Impact factor: 4.123

8.  Gorham-Stout disease in the rib and thoracic spine with spinal injury treated with radiotherapy, zoledronic acid, vitamin D, and propranolol: A case report and literature review.

Authors:  Kazutaka Koto; Kentarou Inui; Megumi Itoi; Kyoko Itoh
Journal:  Mol Clin Oncol       Date:  2019-10-07
  8 in total

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