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 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.
Ozeki
Michio
M
Department of Pediatrics, Gifu University Graduate School of Medicine, Gifu, Japan. mi_ti_ti_1227@yahoo.co.jp
Funato
Michinori
M
Kanda
Kaori
K
Ito
Masahumi
M
Teramoto
Takahide
T
Kaneko
Hideo
H
Fukao
Toshiyuki
T
Kondo
Naomi
N
D002648:Child; D006801:Humans; D000077190:Interferon alpha-2; D016898:Interferon-alpha / Q000008:administration & dosage / Q000097:blood / Q000627:therapeutic use*; D008202:Lymphangioma / Q000000981:diagnostic imaging / Q000188:drug therapy* / Q000503:physiopathology; D008297:Male; D011092:Polyethylene Glycols; D011859:Radiography; D011994:Recombinant Proteins; D042461:Vascular Endothelial Growth Factor A / Q000378:metabolism