CLINICAL IMPROVEMENT OF DIFFUSE LYMPHANGIOMATOSIS WITH PEGYLATED INTERFERON ALFA-2B THERAPY: Case Report and Review of the Literature

Diffuse lymphangiomatosis is a very rare congenital disease, characterized by diffuse or multifocal lymphangioma in the skeletal tissue, spleen, liver, mediastinum, and/or lung. The prognosis is usually poor, especially for children with thoracic lesion, and treatments for the disease are controvers...

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Bibliographic Details
Published in:Pediatric hematology and oncology 2007-01, Vol.24 (7), p.513-524
Main Authors: Ozeki, Michio, Funato, Michinori, Kanda, Kaori, Ito, Masahumi, Teramoto, Takahide, Kaneko, Hideo, Fukao, Toshiyuki, Kondo, Naomi
Format: Article
Language:English
Subjects:
Child
D2-40
diffuse lymphangiomatosis
Humans
Interferon-alpha - administration & dosage
Interferon-alpha - blood
Interferon-alpha - therapeutic use
Lymphangioma - diagnostic imaging
Lymphangioma - drug therapy
Lymphangioma - physiopathology
Male
pegylated interferon alfa-2b
pleural effusion
Polyethylene Glycols
Radiography
Recombinant Proteins
vascular endothelial growth factor
Vascular Endothelial Growth Factor A - metabolism
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Summary:Diffuse lymphangiomatosis is a very rare congenital disease, characterized by diffuse or multifocal lymphangioma in the skeletal tissue, spleen, liver, mediastinum, and/or lung. The prognosis is usually poor, especially for children with thoracic lesion, and treatments for the disease are controversial. The authors report a 9-year-old boy with diffuse lymphangiomatosis involving the thorax with pleural effusions, the spleen, and systemic bone. The patient was treated with pegylated interferon alfa-2b, and achieved good clinical and radiological improvement.
ISSN:0888-0018
1521-0669
DOI:10.1080/08880010701533603