Abstract
Generalized lymphatic malformation (GLM) often causes chylothorax, which can be a
highly morbid condition. Chylothorax in a pediatric patient with a lymphangiogenic
disorder may be especially resistant to therapy, requiring more aggressive measures.
We discuss the complex ongoing long-term management of a pediatric patient with GLM
who presented with massive, persistent left chylothorax, which resolved by surgical
ligation of the thoracic duct and medical management.
Key words
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References
- Chylothorax in Gorham's disease.J Korean Med Sci. 2002; 17: 826-829
- OK-432 therapy for chylous pleural effusion or ascites associated with lymphatic malformations.J Ped Surg. 2010; 45: E7-E10
- Sclerotherapy for lymphangioma in children.Int J Pediatr Otorhinolaryngol. 2003; 67: 795-800
- Lymphatic and venous malformation or “lymphangiohemangioma” of the anterior mediastinum: a case report and literature review.Gen Thorac Cardiovasc Surg. 2011; 59: 575-578
- Progesterone receptors identified in vascular malformations of the head and neck.Otolaryngol Head Neck Surg. 2009; 141: 491-495
- Chylothorax complicating Gorham's disease.Ann Thorac Surg. 2001; 72: 937-939
- Chylothorax in Gorham's syndrome. A common complication of a rare disease.Chest. 1994; 105: 208-213
- Chylothorax: a review of 18 cases.Thorax. 1986; 41: 880-885
- Chylothorax and pseudochylothorax.Clin Chest Med. 1985; 6: 163-171
- Traumatic chylothorax and thoracic duct ligation.J Thorac Surg. 1955; 29: 467-479
- Massive osteolysis: report of two cases.J Bone Joint Surg. 1970; 52-B: 452-459
- Treatment of chylothorax in Gorham's disease: case report and literature review.Can J Surg. 2002; 45: 381-382
- Lymphangiography in the diagnosis and localization of various chyle leaks.Cardiovasc Intervent Radiol. 2012; 35: 117-126
- Lymphoscintigraphy using technetium-99 m HSA-DTPA with SPECT/CT in chylothorax after childbirth.Radiat Med. 2008; 26: 508-511
- Chylothorax associated with massive osteolysis (Gorham's syndrome).Ann Thorac Surg. 2002; 73: 1956-1957
- Gorham's syndrome: a case report and review of the literature.Am J Med. 1987; 83: 1151-1156
- Cyclooxygenase-2 inhibition: effects on tumour growth, cell cycling and lymphangiogenesis in a xenograft model of breast cancer.Br J Cancer. 2007; 96: 575-582
- The selective Cox-2 inhibitor Celecoxib suppresses angiogenesis and growth of secondary bone tumors: an intravital microscopy study in mice.BMC Cancer. 2006; 6: 9
- mTOR-targeted therapy of cancer with rapamycin derivatives.Ann Oncol. 2005; 16: 525-537
- Isolated lymphatic endothelial cells transduce growth, survival and migratory signals via the VEGF-C/D receptor VEGFR-3.EMBO J. 2001; 20: 4762-4773
- Direct recruitment of CRK and GRB2 to VEGFR-3 induces proliferation, migration, and survival of endothelial cells through the activation of ERK, AKT, and JNK pathways.Blood. 2005; 106: 3423-3431
- Sirolimus for the treatment of complicated vascular anomalies in children.Pediatr Blood Cancer. 2011; 57: 1018-1024
Article info
Publication history
Accepted:
January 7,
2013
Received in revised form:
December 21,
2012
Received:
September 3,
2012
Identification
Copyright
© 2013 Elsevier Inc. Published by Elsevier Inc. All rights reserved.
